interventions based on the theory of mind cognitive model for …€¦ · t a b l e o f c o n t e n...

Interventions based on the Theory of Mind cognitive model

for autism spectrum disorder (ASD) (Review)

Fletcher-Watson S, McConnell F, Manola E, McConachie H

This is a reprint of a Cochrane review, prepared and maintained by The Cochrane Collaboration and published in The Cochrane Library2014, Issue 3

http://www.thecochranelibrary.com

Interventions based on the Theory of Mind cognitive model for autism spectrum disorder (ASD) (Review)

Copyright © 2014 The Cochrane Collaboration. Published by John Wiley & Sons, Ltd.

http://www.thecochranelibrary.com

T A B L E O F C O N T E N T S

1HEADER . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

1ABSTRACT . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

2PLAIN LANGUAGE SUMMARY . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

4SUMMARY OF FINDINGS FOR THE MAIN COMPARISON . . . . . . . . . . . . . . . . . . .

8BACKGROUND . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

9OBJECTIVES . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

9METHODS . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

14RESULTS . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

Figure 1. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 15

Figure 2. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 18

Figure 3. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 19

Figure 4. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 22

Figure 5. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 23

Figure 6. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 24

25DISCUSSION . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

27AUTHORS’ CONCLUSIONS . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

27ACKNOWLEDGEMENTS . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

28REFERENCES . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

35CHARACTERISTICS OF STUDIES . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

66DATA AND ANALYSES . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

Analysis 1.1. Comparison 1 Treatment effects in meta-analysis, Outcome 1 Joint engagement in mother-child interaction. 66

Analysis 1.2. Comparison 1 Treatment effects in meta-analysis, Outcome 2 Emotion recognition from face photographs,

TAU control. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 67

Analysis 1.3. Comparison 1 Treatment effects in meta-analysis, Outcome 3 Joint attention initiations in standardised

assessment. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 68

68ADDITIONAL TABLES . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

73APPENDICES . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

78CONTRIBUTIONS OF AUTHORS . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

79DECLARATIONS OF INTEREST . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

79SOURCES OF SUPPORT . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

80DIFFERENCES BETWEEN PROTOCOL AND REVIEW . . . . . . . . . . . . . . . . . . . . .

80INDEX TERMS . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

iInterventions based on the Theory of Mind cognitive model for autism spectrum disorder (ASD) (Review)


[Intervention Review]

Interventions based on the Theory of Mind cognitive modelfor autism spectrum disorder (ASD)

Sue Fletcher-Watson1, Fiona McConnell1, Eirini Manola2 , Helen McConachie3

1Moray House School of Education, University of Edinburgh, Edinburgh, UK. 2Puzzle - School for Children with Autism, Athens,

Greece. 3Institute of Health and Society, Newcastle University, Newcastle upon Tyne, UK

Contact address: Sue Fletcher-Watson, Moray House School of Education, University of Edinburgh, St John’s Land, Holyrood Road,

Edinburgh, EH8 8AQ, UK. [email protected]. [email protected].

Editorial group: Cochrane Developmental, Psychosocial and Learning Problems Group.

Publication status and date: New, published in Issue 3, 2014.

Review content assessed as up-to-date: 7 August 2013.

Citation: Fletcher-Watson S, McConnell F, Manola E, McConachie H. Interventions based on the Theory of Mind cognitive

model for autism spectrum disorder (ASD). Cochrane Database of Systematic Reviews 2014, Issue 3. Art. No.: CD008785. DOI:

10.1002/14651858.CD008785.pub2.


A B S T R A C T

Background

The ’Theory of Mind’ (ToM) model suggests that people with autism spectrum disorder (ASD) have a profound difficulty understanding

the minds of other people - their emotions, feelings, beliefs, and thoughts. As an explanation for some of the characteristic social and

communication behaviours of people with ASD, this model has had a significant influence on research and practice. It implies that

successful interventions to teach ToM could, in turn, have far-reaching effects on behaviours and outcome.

Objectives

To review the efficacy of interventions based on the ToM model for individuals with ASD.

Search methods

In August 2013 we searched CENTRAL, Ovid MEDLINE, Embase, CINAHL, PsycINFO, ERIC, Social Services Abstracts, Autism-

Data, and two trials registers. We also searched the reference lists of relevant papers, contacted authors who work in this field, and

handsearched a number of journals.

Selection criteria

Review studies were selected on the basis that they reported on an applicable intervention (linked to ToM in one of four clearly-defined

ways), presented new randomised controlled trial data, and participants had a confirmed diagnosis of an autism spectrum disorder.

Studies were selected by two review authors independently and a third author arbitrated when necessary.

Data collection and analysis

Risk of bias was evaluated and data were extracted by two review authors independently; a third author arbitrated when necessary. Most

studies were not eligible for meta-analysis, the principal reason being mis-matching methodologies and outcome measures. Three small

meta-analyses were carried out.

1Interventions based on the Theory of Mind cognitive model for autism spectrum disorder (ASD) (Review)


mailto:[email protected]

mailto:[email protected]

Main results

Twenty-two randomised trials were included in the review (N = 695). Studies were highly variable in their country of origin, sample

size, participant age, intervention delivery type, and outcome measures. Risk of bias was variable across categories. There were very few

studies for which there was adequate blinding of participants and personnel, and some were also judged at high risk of bias in blinding

of outcome assessors. There was also evidence of some bias in sequence generation and allocation concealment. Not all studies reported

data that fell within the pre-defined primary outcome categories for the review, instead many studies reported measures which were

intervention-specific (e.g. emotion recognition). The wide range of measures used within each outcome category and the mixed results

from these measures introduced further complexity when interpreting results.

Studies were grouped into four main categories according to intervention target/primary outcome measure. These were: emotion

recognition studies, joint attention and social communication studies, imitation studies, and studies teaching ToM itself. Within the

first two of these categories, a sub-set of studies were deemed suitable for meta-analysis for a limited number of key outcomes.

There was very low quality evidence of a positive effect on measures of communication based on individual results from three studies.

There was low quality evidence from 11 studies reporting mixed results of interventions on measures of social interaction, very low

quality evidence from four studies reporting mixed results on measures of general communication, and very low quality evidence from

four studies reporting mixed results on measures of ToM ability.

The meta-analysis results we were able to generate showed that interventions targeting emotion recognition across age groups and

working with people within the average range of intellectual ability had a positive effect on the target skill, measured by a test using

photographs of faces (mean increase of 0.75 points, 95% confidence interval (CI) 0.22 to 1.29 points, Z = 2.75, P < 0.006, four studies,

N = 105). Therapist-led joint attention interventions can promote production of more joint attention behaviours within adult-child

interaction (mean increase of 0.55 points, 95% CI 0.11 to 0.99 points, Z = 2.45, P value = 0.01, two studies, N = 88). Further analysis

undermines this conclusion somewhat by demonstrating that there was no clear evidence that intervention can have an effect on joint

attention initiations as measured using a standardised assessment tool (mean increase of 0.23 points, 95% CI -0.48 to 0.94 points, Z

= 0.63, P value = 0.53, three studies, N = 92). No adverse effects were apparent.

Authors’ conclusions

While there is some evidence that ToM, or a precursor skill, can be taught to people with ASD, there is little evidence of maintenance

of that skill, generalisation to other settings, or developmental effects on related skills. Furthermore, inconsistency in findings and

measurement means that evidence has been graded of ’very low’ or ’low’ quality and we cannot be confident that suggestions of positive

effects will be sustained as high-quality evidence accumulates. Further longitudinal designs and larger samples are needed to help

elucidate both the efficacy of ToM-linked interventions and the explanatory value of the ToM model itself. It is possible that the

continuing refinement of the ToM model will lead to better interventions which have a greater impact on development than those

investigated to date.

P L A I N L A N G U A G E S U M M A R Y

A review of evidence on the use of interventions for people with autism spectrum disorder, based on the psychological model

’Theory of Mind’

Background

The ’Theory of Mind’ model suggests that people with autism spectrum disorder (ASD) have a profound difficulty understanding

the minds of other people, their emotions, feelings, beliefs, and thoughts. It has been proposed that this may underlie many of the

other difficulties experienced by people with ASD, including social and communication problems, and some challenging behaviours.

Therefore, a number of studies have attempted to teach theory of mind and related skills to people with ASD.

Review question

This review aimed to explore whether a) it is possible to teach theory of mind skills to people with autism and b) whether or not this

evidence supports the theory of mind model. Having a ’theory of mind’ may depend on developing related basic skills, including joint

attention (sharing a focus of interest with another person), recognising other people’s emotions from faces or stories, and imitating

other people. Therefore, we included intervention studies that taught not just theory of mind itself, but also related skills.

Study characteristics



We found 22 research studies involving 695 participants, which reported on the efficacy of interventions related to theory of mind.

The evidence is current to 7th August 2013.

Key results and the quality of the evidence

Despite all studies using a high-quality basic methodology (the randomised controlled trial), there was concern over poor study design

and reporting in some aspects. While there is some evidence that theory of mind, or related skills, can be taught to people with ASD,

there is currently poor quality evidence that these skills can be maintained, generalised to other settings, or that teaching theory of

mind has an impact on developmentally-linked abilities. For example, it was rare for a taught skill to generalise to a new context, such

as sharing attention with a new adult who was not the therapist during the intervention. New skills were not necessarily maintained

over time. This evidence could imply that the theory of mind model has little relevance for educational and clinical practice in ASD.

Further research using longitudinal methods, better outcome measures, and higher standards of reporting is needed to throw light on

the issues. This is particularly important as the specific details of the theory of mind model continue to evolve.



S U M M A R Y O F F I N D I N G S F O R T H E M A I N C O M P A R I S O N [Explanation]

Theory of Mind based interventions compared with wait-list or treatment-as-usual control for autism spectrum disorder.

Patient or population: People with autism spectrum disorder

Settings: Schools, home and clinical settings

Intervention: Based on the Theory of Mind theoretical model of autism

Comparison: Most studies incorporate an ’empty’ control such as treatment-as-usual or wait-list

Outcomes Illustrative comparative risks* (95% CI) No of Participants

(studies)

Quality of the evidence

(GRADE)

Comments

Assumed risk Corresponding risk

[Control] [Intervention]

Symptom Level: Communica-

tion

Various measures, including:

Autism Diagnostic Observa-

tion Schedule (ADOS)

Conversation Skills

Social Communication Ques-

tionnaire (SCQ) (level of eye-

contact)

See ’Corresponding Risk’ Wong 2010 and Young 2012

report positive effects of in-

tervention on symptom level

in the communication domain,

while Hadwin 1996 found no

effect on conversational skills

(this specific outcome is re-

ported in Hadwin 1997)

ADOS: n = 17

(Wong 2010)

Conversation: n = 30

(Hadwin 1996)

SCQ: n = 25

(Young 2012)

⊕©©©

very low [1]

Three included studies report

outcomes in this area of clin-

ical relevance. Each one uses

a different assessment to cap-

ture change in this domain.

One study uses an unstan-

dardised measure, though it

is designed to capture change

over time Hadwin 1996). The

other two studies use stan-

dardised measures of com-

munication skills but neither of

these were designed to cap-

ture change over time nor to

be used as intervention out-

come measures

Symptom Level: Social Inter-

action


Autism Diagnostic Observa-

tion Schedule (ADOS)

See ’Corresponding Risk’ Fewer than half of the rele-

vant included studies report

positive effects of interven-

tion on symptom level in

the social interaction domain

ADOS: n = 17

(Wong 2010)

CSBS: n = 48

(Landa 2011)

ESCS: n = 200

⊕⊕©©

low [2]

Here we include both stan-

dardised assessments and di-

rect observations of social be-

haviours

Eleven included studies report

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http://www.thecochranelibrary.com/view/0/SummaryFindings.html

Communication and Symbolic

Behaviour Scale (CSBS)

Early Social Communication

Scales (ESCS)

PDD-BI social approach sub-

scale

Precursors of Joint Attention

Measure

Social (PJAM) Communica-

tion Questionnaire (SCQ)

Social Emotional Scale (SES)

(Bayley-III)

Social Skills Rating System

(SSRS)

Vineland Adaptive Behaviour

Scales (socialisation) (VABS)

andOther social interaction (SI)

observations

(Ingersoll 2012; Kasari 2006;

Hopkins 2011; Landa 2011;

Wong 2010).

In addition some studies re-

port mixed findings across

methods. For example, Goods

2013 and Kaale 2012 report

some positive effects mea-

sured in observations, but null

findings from the ESCS. Con-

versely Kim 2009 (outcomes

reported in Kim 2008) and

Wong 2013 find significant ef-

fects measured by the ESCS

but not all other measures. In

the case of Wong 2013 this

is further complicated by a

mixed output from the ESCS

where a significant effect is

found for one scored item but

not another. Similar findings

are reported by Schertz 2013

and Kasari 2010 who find

positive effects of intervention

on some observed behaviours

but not others

Both studies which report the

impact of an emotion recog-

nition intervention on gener-

alised social skills do not

find significant effects on their

chosen outcomes (Williams

2012; Young 2012)

(Goods 2013; Ingersoll 2012;

Kaale 2012; Kasari 2006; Kim

2009; Wong 2013)

PDD-BI: n = 10

(Kim 2009)

PJAM: n = 23

(Schertz 2013)

SCQ: n = 25

(Young 2012)

SES Bayley: n = 27

(Ingersoll 2012)

SSRS: n = 49

(Hopkins 2011)

VABS Socialisation: n = 55

(Williams 2012)

Other SI: n = 175

(Kasari 2006, Kim2009; Kaale

2012; Kasari 2010; Goods

2013)

outcomes in this area of clin-

ical relevance. There is wide

variety in the choice of as-

sessments to capture change

in this domain, though most

are based on standardised as-

sessments and are often de-

signed to capture change over

time

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General Communication Abil-

ity (e.g. vocabulary)

Mullen Scales of Early Learn-

ing (MSEL)

Reynell Developmental Lan-

guage Scales

See ’Corresponding Risk’ Schertz 2013 reports signifi-

cant intervention effects on re-

ceptive language and a non-

significant but moderate sized

effect (d = 0.78) for ex-

pressive language scores. At

a one-year follow-up Kasari

2006 likewise report interven-

tion effects on expressive lan-

guage, which were signifi-

cantly greater for the joint at-

tention intervention compared

with both control group and

symbolic play interventions.

However these effects on

expressive vocabulary were

not sustained four years later

(Kasari 2012b). In addition, a

methodologically strong study

(Landa 2011) reports no ef-

fects on expressive language.

MSEL expressive: n = 71

(Landa 2011, Schertz 2013)

MSEL receptive:

n = 23

(Schertz 2013)

Reynell: n = 58

(Kasari 2006)

⊕©©©

very low [3]

Though this has commonly

been used as an outcome

measure in generalised social

skills interventions for children

with ASD, only three of the

studies included in this review

report a general communica-

tion ability outcome measure

Theory of Mind ability


False-belief tasks

Happe’s Strange Stories

Faux-Pas Recognition Test

NEPSY-II ToM tasks

The ToM Test

See ’Corresponding Risk’ Two studies report some pos-

itive effects of intervention

on ToM ability (Begeer 2011;

Fisher 2005) one reports no

impact on directly-assessed

ToM ability (Solomon 2004)

and one reports a reduction at

follow-up in ToM ability for the

intervention group specifically

(Williams 2012).

False belief: n = 27

(Fisher 2005)

Happe SS, & Faux-Pas RT: n

= 18

(Solomon 2004)

NEPSY: n = 55

(Williams 2012)

ToM Test: n = 36

(Begeer 2011)

⊕©©©

very low

[1]

Four included studies report

outcomes in this area of prin-

cipally theoretical relevance.

There is wide variety in the

choice of assessments to

capture change in this do-

main, though most are based

on standardised assessments

and are closely linked to the

intervention target skill

*The basis for the assumed risk (e.g. the median control group risk across studies) is provided in footnotes. The corresponding risk (and its 95% confidence interval) is based on the

assumed risk in the comparison group and the relative effect of the intervention (and its 95% CI).

CI: Confidence interval; RR: Risk Ratio

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GRADE Working Group grades of evidence

High quality: Further research is very unlikely to change our confidence in the estimate of effect.

Moderate quality: Further research is likely to have an important impact on our confidence in the estimate of effect and may change the estimate.

Low quality: Further research is very likely to have an important impact on our confidence in the estimate of effect and is likely to change the estimate.

Very low quality: We are very uncertain about the estimate.

1. Risk of bias (-1); inconsistency (-2): Since the studies included here are of variable methodological quality and report mixed findings

this evidence is considered to be of Very Low quality.

2. Risk of bias (-1); inconsistency (-1): The studies included here are of variable methodological quality and report mixed findings from

a wide variety of measures. There is a collection of studies reporting on the ESCS (some of which are summarised in Analysis 1.1), but

within this group findings are once again mixed. Indeed, even within a single study and measure there may be inconsistency in evidence

for intervention efficacy. It is therefore impossible to be confident about the impact of Theory of Mind interventions on social interaction

domain symptom level and the evidence quality is rated as Low.

3. Risk of bias (-1); inconsistency (-1); low sample size (-1): These mixed outcomes from only a handful of studies must be judged of

Very Low Quality until they can be resolved by additional high-quality evidence.

It is challenging to divide communication and social interaction for measures which tap into both of these qualities. However for the

purposes of this table, we have identified measures which are based on observation of an interpersonal interaction as falling into the

Social Interaction Domain.

A number of included studies report on measures of emotion recognition and imitation skill. While these are suitable outcomes for the

respective interventions, and highly associated with ASD profiles, these cannot be categorised into the domains for this Summary of

Findings table, and therefore are not addressed here.

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B A C K G R O U N D

Description of the condition

Autism spectrum disorder (ASD) is an umbrella term used to de-

scribe all people diagnosed as showing symptoms within two core

criteria: communication and social deficits, and fixed or repeti-

tive behaviours (APA 2013). The ASD label replaces former sub-

types, including autism, pervasive developmental disorder - not

otherwise specified (PDD-NOS), and Asperger’s syndrome (AS)

(APA 1994). Likewise, the single communication and social in-

teraction cluster is derived from what was originally two separate

domains of impairment in communication and social interaction

(APA 1994). These difficulties often make it very hard for people

with ASD to be successful members of society and can present very

serious challenges to parents, teachers, and other professionals.

Prevalence estimates of ASD diagnosis in children have been rising

significantly in recent years with an authoritative systematic review

estimating global prevalence of pervasive developmental disorders

at 62 per 10,000 and autistic disorder at 17 per 10,000 (Elsabbagh

2012). Figures may be higher in more developed countries (e.g.

Baird 2006). This represents a more-than-threefold increase on

previously published figures, which estimated autism prevalence

at about 5 per 10,000 (Fombonne 2001). While there are method-

ological differences between prevalence studies, the rising preva-

lence of ASD has been well-documented across Western countries,

including Europe, Australia, and the USA (e.g. Yeargin-Allsopp

2003; Williams 2006; Atladottir 2007; Kogan 2009; Nassar 2009).

There has been significant debate about the cause of the recent

rise in prevalence of ASD, but the influence of increased awareness

of the disorder among health professionals and the community

at large, and the role of diagnostic substitution, should not be

underestimated (Croen 2002; Atladottir 2007). There are other

candidate explanations, including the possibility of environmental

causes of the rising prevalence estimates, though, as yet, there

is no good empirical evidence for these (Rutter 2005). Baird et

al (Baird 2006) conclude that “Whether the increase is due to

better ascertainment, broadening diagnostic criteria, or increased

incidence is unclear” (p. 210).

Within the disorder there is a male to female ratio of 4:1 or 5:

1 (Baird 2006; Kogan 2009), as noted in the set of case stud-

ies, which defined the condition for the first time (Kanner 1943).

ASDs have this feature in common with most other neurodevelop-

mental disorders (such as attention deficit hyperactivity disorder

(ADHD), dyslexia, dyspraxia), though to a greater extent. As yet,

there is no empirical evidence for systematic differences between

male and female individuals with ASD (Hartley 2009).

Theory of Mind

The term ’Theory of Mind’ (ToM) describes the ability to un-

derstand another’s thoughts, beliefs, and other internal states and

was originally applied to the study of non-human primate cog-

nition (Premack 1978). The term has since been developed in a

number of different directions (e.g. Carruthers 1996), including

in research into ASD. The first application of the term in ASD

research was in an experiment which used false-belief paradigms

to explore ToM in children with autism (Baron-Cohen 1985). In

this study, children were presented with a scenario in which a doll,

Sally, ’believed’ her marble was in the basket where she left it.

However, the child and experimenter knew that while Sally was

elsewhere, another doll had moved the marble into a box. The key

question was “Where will Sally look for her marble?” Typically-

developing children from the age of four years, sometimes earlier,

can correctly ascertain that Sally will look in the basket; she holds

a false belief about the location of the marble (Wellman 2001).

Children with ASD are much less likely to give a correct answer

to this question at age four years. They normally claim that Sally

will look in the box, in accordance with reality, but incompatible

with Sally’s knowledge of the situation.

Research into ToM in children and adults with ASD has been

prolific over the last 25 years (e.g. Baron-Cohen 2000). While the

details are subject to debate, it is widely accepted that people with

ASD do not possess a fully-functioning theory of mind; even high-

functioning adults with ASD may struggle with complex ToM

tasks (Ponnet 2004). ToM has been placed in a developmental

context, consisting of a range of precursor skills, including fol-

lowing eye-gaze, establishing joint attention, imitation, pretend

play, and emotion recognition (Melzoff 1993; Baron-Cohen 1995;

Charman 2000; Wellman 2000; Ruffman 2001). ToM then also

links to subsequent social and communication skills, including the

development of language (Tager-Flusberg 2000; Garfield 2001).

As a result, many believe that failures of ToM are central to ex-

plaining the difficulties experienced by people with ASD (though

not a sufficient explanation). Therefore, ToM and its precursor

skills are targets for interventions.

Description of the intervention

A ’Theory of Mind intervention’ is a treatment or therapy, which

is explicitly or implicitly based on the Theory of Mind (ToM)

cognitive model of ASD. ToM interventions target those skills

which are either potential components or precursors of ToM (

Swettenham 2000). One example of an intervention targeting such

skills is using ’thought-bubbles’ to teach children with ASD to

understand others’ thoughts and beliefs by illustrating these in

bubbles (as in a cartoon) (Parsons 1999). Specific precursor skills

can also be taught such as helping a child to make eye-contact

to accompany pointing to an object of interest (joint attention).

More detail on which interventions are eligible for inclusion in this

review is given in the Methods section, but we will only consider

interventions that explicitly target ToM skills.

ToM interventions can be contrasted with other types of treat-

ment for ASD. Many intervention models focus on behaviour



management and personal skills training, using a basic condition-

ing model for learning (repetition; rewarding desirable behaviour;

’punishing’ or ignoring behaviour that the therapist finds undesir-

able such as tantrums). In addition, most management strategies

for ASD occur within a fairly structured timetable as people with

ASD tend to feel more comfortable following familiar routines in

a consistent environment, and respond very poorly to change.

How the intervention might work

In a chapter reviewing evidence for the possibility of teaching ToM

to individuals with autism, Swettenham states (p. 442) that “a

successful method for teaching theory of mind may alleviate the

impairments in social interaction that are so debilitating in autism”

(Swettenham 2000).

The ToM model of autism suggests that the social and commu-

nication difficulties that are characteristic of the syndrome stem

from a failure to develop an intact ToM. Certainly there is evidence

that ToM is correlated with real-life social skills (Frith 1994) and

symptomatology (Joseph 2004). Certain ToM precursor skills also

have a direct relationship with symptoms (Mundy 1994). There-

fore, training in ToM, or in the precursor or component skills of

ToM, should alleviate the social and communication difficulties

experienced by individuals with the disorder. For example, a tar-

geted joint attention intervention for autism produced improve-

ments in children’s responsiveness to joint attention opportuni-

ties and also improved sharing and language (Kasari 2006; Kasari

2008), indicating that ToM interventions may have consequences

for wider developmental abilities.

It is possible that interventions targeting different ToM skills will

produce varied types of change in participants, and the extent of

change may vary. The method of delivery of the intervention may

also produce different outcomes. For example, one might expect an

intervention delivered by a trained therapist to have greater impact

than one delivered by parents. An intervention taught in school

may have a different impact to one delivered in the home. The

duration of the intervention may also be significant. Deficits in

ToM and related skills vary with age (Happe 1995), IQ (Ozonoff

1991a; Happe 1994; Bowler 1997), specific diagnosis (Ozonoff

1991b; Bowler 1992) and verbal ability (Happe 1995; Garfield

2001). As a result, the specific skill being targeted, the method of

intervention delivery, its duration and individual differences be-

tween participants in ToM intervention studies will be important

factors for consideration and for statistical analysis in this review.

Why it is important to do this review

To date, there is no comprehensive review of ToM interventions

for autism, despite the fact that the first study attempting to teach

ToM to individuals with autism was published in 1995 (Ozonoff

1995). This review will be of relevance to both the clinical and

academic research communities, since ToM interventions not only

have the potential to benefit people with ASD, but also provide a

unique and rigorous way to test the theoretical model on which

they are based.

O B J E C T I V E S

To assess the effect of interventions, based on the Theory of Mind

(ToM) model, for autism spectrum disorders (ASD), on symp-

toms in the core diagnostic domains of social and communication

impairments in autism, and on language and ToM skills. In addi-

tion, in so-doing, to test the applied value of the ToM model of

autism.

M E T H O D S

Criteria for considering studies for this review

Types of studies

Randomised and quasi-randomised trials (defined as trials in

which allocation was made by, for example, alternate allocation or

allocation by date of birth).

Types of participants

Participants of any age with a diagnosis of an ASD, including

autism, atypical autism, Asperger’s syndrome, and PDD-NOS,

according to either ICD-10 (Internal Classification of Diseases),

DSM-IV or DSM-V (Diagnostic Statistical Manual of Mental

Disorders) criteria. All diagnostic categories could be included

since the validity of differentiating between categories on the spec-

trum is not well established (Klin 2005). Furthermore, the ToM

cognitive model does not distinguish, on a qualitative basis, be-

tween different forms of ASD. Participants must have received a

‘best estimate’ clinical diagnosis, confirmed by the study authors.

That is, at a minimum, diagnosis by a multidisciplinary clinical

team using standard procedures with reference to the international

classification systems. Use of a particular diagnostic tool, such

as the Autism Diagnostic Observation Schedule (ADOS) (Lord

2000) or the Autism Diagnostic Interview (ADI-R) (Lord 1994),

was desirable but not required. Co-morbid cases were also eligible

for inclusion since these individuals are just as needful of inter-

vention for their specifically autistic difficulties.



Types of interventions

Interventions eligible for inclusion in this review:

1. explicitly state that they are designed to teach ToM; or

2. explicitly state that they are designed to teach precursor

skills of ToM; or

3. explicitly state that they are based on or inspired by ToM

models of autism; or

4. explicitly state that they aim to test the ToM model of

autism.

We reiterate that ToM (theory of mind) describes the ability to

understand another’s thoughts, beliefs, and other internal states

and is encapsulated in a test of false belief. Prior to the develop-

ment of false-belief understanding (at about four years old in typ-

ical development), associated precursor skills are in evidence such

as joint attention, imitation, and emotion recognition. Relevant

interventions include those which explicitly teach children to un-

derstand others’ mental states (e.g. using visual representations of

mental states McGregor 1998) and those which use naturalistic

teaching to develop imitation skills (Heimann 2006).

The following kinds of interventions are not included in this re-

view:

1. interventions which do not meet the criteria given above;

2. medical interventions (e.g. risperidone for aggression in

ASD);

3. dietary interventions (e.g. gluten-free and casein-free diets);

4. interventions which target a particular behaviour rather

than a cognitive skill (e.g. over-sensitivity to light modified using

colour spectacles; sleep difficulties modified using applied

behavioural analysis);

5. language-focused interventions (e.g. to make requests using

the Picture Exchange Communication System or spoken single

words);

6. interventions which have a broad-base both in terms of

methods (e.g. combining computerised learning with parent

training and social skills groups) and targets (i.e. addressing a

range of social communication skills, some which are ToM-

linked but also more general skills such as turn-taking, friendship

skills, and conversation).

ToM interventions are compared with the following conditions,

where these are used:

1. treatment-as-usual/wait-list control;

2. ‘placebo’ interventions, for example a ‘contact control’ such

as watching Thomas the Tank Engine DVDs (e.g. Young 2012);

3. intervention with no therapeutic content, (e.g. group

leisure activities (Baghdadli 2013).

All ‘doses’ (that is the number and length of treatment sessions per

week), durations, and methods were eligible for inclusion.

Types of outcome measures

Outcome measures do not form part of the criteria for inclusion

of studies in this review.

Primary outcomes

Primary outcomes at a participant symptom level, measured using

standardised diagnostic assessments or clinical report. Outcomes

will be in each of two symptom domains that have until recently

been used in clinical diagnosis and are followed by most diagnostic

tests for autism. These are as follows, with examples of outcomes

in each category as measured by the ADOS (Lord 2000) or ADI

(Lord 1994).

1. Communication: overall level of non-echoed language;

stereotyped or idiosyncratic use of words or phrases; pointing;

gestures; conversation.

2. Social function: unusual eye-contact; facial expressions

directed to others; spontaneous initiation of joint attention;

shared enjoyment in interaction; quality of rapport.

The third diagnostic domain of Restricted and Repetitive Be-

haviours (imaginative play or creativity; unusual sensory interests;

unusually repetitive interests or stereotyped behaviours; compul-

sions or rituals) is not included as an expected primary outcome.

Secondary outcomes

In addition, the following secondary outcomes will be included.

PARTICIPANT, direct measurement

• Intervention-specific: change in targeted cognitive skill such

as false-belief understanding

• Change in participant behaviour or quality of interpersonal

interaction, or both, measured by direct observation.

PARENT, teacher (or other individual in caring or educational

relationship to the participant) report

• Change in participant behaviour and skills or deficits such

as: adaptive skills; school success; challenging behaviours; social

participation measured by parent, teacher or other report

• Acceptability of intervention (time, cost)

OTHER

• Intervention process measures e.g. rate of drop-out

• Economic data e.g. financial cost of intervention; time

commitment required

Main outcomes for ’Summary of findings’ table

The following outcomes measures are specified for a ’Summary of

findings’ table:

1. symptom level, communication domain;

2. symptom level, social interaction domain;

3. general communication ability (e.g. vocabulary);

4. ’Theory of Mind’ ability (e.g. false-belief test score).

Where data were available, we planned to organise outcomes into

three time points: immediately post-treatment; medium-term out-

come (up to six months post-treatment); and long term (more

than six months post-treatment).

The Summary of findings for the main comparison reports on

these outcomes and also includes an estimate of the quality of

evidence in each category.



Search methods for identification of studies

The complex nature of ToM interventions makes them difficult to

capture adequately using search terms. Therefore, to avoid miss-

ing relevant studies, we used a highly sensitive search strategy with

just two concepts: the condition (ASD) and a search filter to find

RCTs. The core search strategy was developed in Ovid MED-

LINE and uses the Cochrane highly sensitive search strategy for

identifying randomised trials (Lefebvre 2008), The MEDLINE

strategy was adapted for other databases using appropriate syntax

and controlled vocabulary. The initial searches were run in July

2010 without any date or language restrictions. We last updated

the searches on 6 August 2013, apart from ASSIA which was no

longer available to us.

Electronic searches

We searched the following databases in August 2013.

• Cochrane Central Register of Controlled Trials

(CENTRAL) 2013, Issue 7, part of The Cochrane Library.

• Ovid MEDLINE(R) 1946 to July Week 4 2013.

• EMBASE 1980 to 2013 Week 31.

• CINAHLPlus 1937 to current.

• PsycINFO 1806 to July Week 5 2013.

• ERIC 1966 to current.

• Applied Social Sciences Index and Abstracts: ASSIA (CSA)

1987 to current.

• Social Services Abstracts 1979 to current.

• metaRegister of Controlled Trials (controlled-trials.com/

mrct/).

• ICTRP (apps.who.int/trialsearch/).

• UKCRN - UK Clinical Trials Network (

public.ukcrn.org.uk/search/).

• ClinicalTrials.gov (clinicaltrials.gov/).

• Autism Data (autism.org.uk/autismdata/).

The search strategies for each source are in Appendix 1.

Searching other resources

In addition to searches of electronic databases, we contacted key

authors in the field directly and asked them to provide any rele-

vant published, unpublished or in-progress data, including post-

graduate dissertations. We also searched the bibliographies of key

articles for citations of papers not found electronically. Searches

were made for in-progress, or unpublished clinical trials. Finally,

we searched the online databases of journals that regularly publish

work on this topic. These journals were the Journal of Autism andDevelopmental Disorders, Journal of Child Psychology and Psychiatry,and Autism: International Journal of Research and Practice. We also

searched the proceedings of the International Meeting for AutismResearch.

Data collection and analysis

Selection of studies

All citations sourced from the search strategy were transferred to

EndNote, a reference management programme. Initial screening

of titles and abstracts by an experienced research assistant (EM or

FMcC) eliminated all those citations obviously irrelevant to the

topic, for example, prevalence studies, studies unrelated to ASD,

and single case studies. Thereafter, two review authors (SFW and

either EM or FMcC) assessed and selected studies for inclusion

from the group of superficially relevant studies. In the event of a

disagreement, resolution was reached in discussion with a third

author (HM), if necessary following inspection of the full paper.

Data extraction and management

Two review authors (SFW and either EM or FMcC) indepen-

dently extracted data from selected trials using a specially designed

data extraction form. Extracted data included methods (dose and

frequency of intervention); diagnostic description of participants,

and type of intervention, including target, intensity, duration, and

method of application (parent-mediated, therapist, school-based

etc.). Disagreements were resolved in consultation with a third

author (HM).

Assessment of risk of bias in included studies

Two review authors (SFW and either EM or FMcC) indepen-

dently assessed the risk of bias of included studies in the following

domains: sequence generation; allocation concealment; blinding

of participants and personnel; blinding of outcome assessment; in-

complete outcome data; selective outcome reporting; other sources

of bias. We used The Cochrane Collaboration tool for assessing

risk of bias in these areas. The process involved recording the ap-

propriate information for each study (e.g. describing the method

used to conceal allocation in detail) and evaluating whether there

was risk of bias in that area (e.g. was allocation adequately con-

cealed?). Any disagreement was resolved by referral to a third au-

thor (HM).

We contacted authors to supply missing information from 16 in-

cluded studies (Bolte 2002; Solomon 2004; Fisher 2005; Golan

2006; Kasari 2006; Kim 2009; Golan 2010; Kasari 2010; Ryan

2010; Wong 2010; Hopkins 2011; Ingersoll 2012; Young 2012;

Baghdadli 2013; Schertz 2013; Wong 2013) and information

was received from the majority of authors with the exception of

(Solomon 2004; Fisher 2005; Golan 2006; Golan 2010; Wong

2010; Baghdadli 2013).

Studies were allocated to categories according to our evaluation of

each area or potential risk of bias as follows:



http://www.controlled-trials.com/mrct/



http://apps.who.int/trialsearch/

http://apps.who.int/trialsearch/

http://public.ukcrn.org.uk/search/

http://public.ukcrn.org.uk/search/

http://clinicaltrials.gov/

http://www.autism.org.uk/autismdata/

http://www.autism.org.uk/autismdata/

Random sequence generation

Low risk of bias: adequate sequence generation as indicated by

reference to, e.g., random number table, coin tossing, shuffled

cards or envelopes, throwing dice, drawing lots.

Unclear (or moderate) risk of bias: indicates uncertainty about

whether the sequence was randomly generated.

High risk of bias: a non-random component is described such as

sequence generation by odd or even date of birth, by geographical

location or by date of entry to the study.

Allocation concealment

Low risk of bias: participants and investigators enrolling partici-

pants unable to foresee assignment as indicated by reference to,

e.g., central allocation, opaque envelope procedure, allocation by

an independent partner outside the research team.


whether the allocation was concealed.

High risk of bias: participants and investigators enrolling partici-

pants may have been able to foresee assignment as indicated by ref-

erence to an open random allocation schedule (e.g. random num-

bers list), unsealed or non-opaque envelopes, alternate allocation,

allocation by non-random criteria such as date of birth.

Blinding of participants and personnel

Low risk of bias: participants and personnel blinded to study hy-

potheses and treatment condition, or incomplete blinding but au-

thors judge that outcome is unlikely to be influenced by lack of

blinding. Examples might be when participants are very young

and/or low-functioning people with autism and are unlikely to be

aware of intervention targets, and where outcome is assessed us-

ing a measure resilient to performance bias such as computerised

assessment. We note that in behavioural studies, such as those in-

cluded in this review, it is rarely possible to blind participants and/

or personnel.


whether blinding was consistent, perhaps due to insufficient in-

formation being available, or partial blinding (e.g. of participants

but not personnel).

High risk of bias: participants and personnel not blind to study

hypotheses or treatment condition, and outcome likely to be in-

fluenced by this lack of blinding.

Blinding of outcome assessment

Low risk of bias: outcome examiners and scorers blinded to partic-

ipant group membership, or blinding of some outcome assessors

with good evidence of agreement between blinded and unblinded

raters on outcome measures, or outcome assessors not blind but

outcome measurement unlikely to be influenced by this lack of

blinding.


whether blinding was consistent, perhaps due to insufficient in-

formation being available.

High risk of bias: outcome examiners and scorers not blind to par-

ticipant group membership, and outcome likely to be influenced

by this lack of blinding.

Incomplete outcome data

Low risk: no missing data, or reasons for missing outcome data

unlikely to be related to true outcome, or missing data balanced

across groups with similar reasons in each case.

Unclear risk of bias: insufficient reporting of attrition or exclusions

to permit accurate judgement.

High risk of bias: reasons for missing data likely to be related to true

outcome, with imbalance in numbers between groups or different

reasons between groups.

Selective reporting

Low risk: study protocol available and all pre-specified outcomes

are reported in the pre-specified way, or clear from the published

reports that all expected outcomes are included.

Unclear (moderate) risk of bias: insufficient information to permit

accurate judgement.

High risk of bias: not all of the study’s primary outcomes have been

reported, or outcomes which were not pre-specified are reported,

or one or more primary outcomes have been reported for only

a sub-set of the sample, or one or more outcomes are reported

incompletely so that they cannot be entered into a meta-analysis.

Other bias

Low risk of bias: the study appears to be free of other sources of

bias.

Unclear (or moderate) risk of bias: there may be an additional risk

of bias but there is insufficient information to fully assess this risk,

or it is unclear whether the risk would introduce bias in study

results.

High risk of bias: the study has one important additional risk of

bias such as a source of bias related to the study design, or claims

of fraudulence.

In each case, only studies where the assessment of overall risk falls

into categories ’Low’ or ’Unclear/Moderate’ have been included

in subsequent analyses.

Measures of treatment effect

Binary and categorical data

No studies reported binary outcome data in the current review

version. Should they be included in future updates, methods for



analysing them appear in the published protocol for the review

(Fletcher-Watson 2010).

Continuous data

Where standardised assessment tools generated a continuous score

as the outcome measure, and means and standard deviations were

reported or provided by the authors, comparisons were made be-

tween the means of these scores. When selecting studies for pos-

sible meta-analysis the following criteria were of principal impor-

tance.

• Similarity of trial design - especially whether the ’control’

condition had therapeutic content or not

• Similarity of intervention target

• Similarity of outcome measure - the quality being measured,

the unit of measurement, and the method of measurement (e.g.

parent-report, video coding, standardised assessment)

Where measures were on different scales but those scales were clin-

ically homogeneous, meta-analyses used standardised mean differ-

ence with Hedges’ g correction for small sample sizes.

Unit of analysis issues

No cluster-randomised trials were included in the current review

version. Methods for analysis are recorded in additional Table 1.

Dealing with missing data

Missing data were assessed for each individual study according to

the reports provided by authors. For included studies reporting

drop-out, we reported the number of participants included in the

final analysis as a proportion of those participants who began the

intervention (see Characteristics of included studies). Reasons for

missing data are also reported (that is, whether data are missing at

random or not). In all cases, we concluded that data were missing

at random, and the remaining data were analysed and the missing

data ignored.

Where summary data are missing, trial authors were contacted.

If no reply was forthcoming or the required summaries were not

made available, the study was included in the review and we as-

sessed and discussed the extent to which its absence from meta-

analysis affects the review results (e.g. Bolte 2002).

No studies reported the loss of significant quantities of data, with-

out sufficient explanation, and there was no evidence of non-ran-

dom missing data. Therefore, the review authors agree that the

conclusions of individual studies are not compromised by miss-

ing data. The extent to which the results of the review may be

altered by the missing data is assessed and discussed (Quality of

the evidence).

Additional procedures for dealing with non-random missing data

in future appear in the published review protocol (Fletcher-Watson

2010) and Table 1.

Assessment of heterogeneity

Consistency of results was assessed visually and by a Chi2 test.

Where meta-analysis included only a small number of studies, or

where studies had small sample sizes, a P value of 0.10 was applied

for statistical significance. In addition, since Chi2 can have low

power when only a few studies or studies of a small sample size

are available, we used the I2 statistic to measure the amount of

observed variability in effect sizes that can be attributed to true

heterogeneity (Higgins 2008).

Assessment of reporting biases

Where sufficient studies were found, funnel plots were inspected

to investigate any relationship between effect size and sample size.

Such a relationship could be due to publication or related biases,

or due to systematic differences between small and large studies.

Data synthesis

Data synthesis was performed using RevMan 5.2. Binary data

were not reported in any of the included studies but could be

assessed in future review versions. Where two or more studies

suitable for inclusion were found, and the studies were considered

to be homogenous, a meta-analysis was performed on the results.

Homogeneity decisions were based on examination of a series of

factors identified in the review protocol including the following.

• Similarity in intervention delivery type (e.g. therapist-led,

parent training)

• Similarity in intervention target skill (e.g. emotion

recognition, imitation, joint attention)

• Similarity in participant populations (e.g. intellectual level

in the normal or low range, specific autism diagnostic category,

age)

In addition, the following two further factors were developed post

hoc in response to the wide variability in study design and outcome

measure found.

• Similarity in primary outcome measurement

• Similarity in comparison group status (e.g. did the study

compare two different interventions or compare an intervention

with a wait-list or treatment-as-usual control)

It is essential to distinguish between measures of primary outcome

when assessing intervention efficacy for two main reasons. The first

is that there is significant evidence that people with ASD do not

generalise skills across contexts. For example, Golan 2010 found

differences in outcome measures by close and distant generalisa-

tion tasks even though these were all measures of emotion and

mental state recognition. Therefore, studies measuring outcome

using tasks which differ in complexity and in connection to the

teaching context should not be compared directly. The second rea-

son is that the method of measurement can produce widely varying

distributions, which are not amendable to combination. For exam-

ple, Kasari 2010 measured percentage of total time that a mother-



child dyad were jointly engaged, while Goods 2013 reported the

number of instances that specific types of joint engagement were

observed. It is not possible to combine these two variables, which

have also been collected in different settings (laboratory artificial

mother-child play versus naturalistic classroom observation) and

over different periods of time.

Comparison group status is another key consideration when com-

bining studies. A study that shows an intervention effect compared

with a ’placebo’ group or compared with another intervention may

have a smaller effect size than one comparing intervention and

wait-list control. However, the former study has the more power-

ful design and so this smaller effect should be more influential on

conclusions.

A random-effects model analysis was used since we do not assume

that each study is estimating exactly the same quantity.

Subgroup analysis and investigation of heterogeneity

Subgroup analyses were not possible in this version of the review.

Dimensions for possible future subgroup analyses are included in

additional Table 1.

Sensitivity analysis

Sensitivity analysis was not possible for this version of the review.

Details of planned future sensitivity analyses are included in addi-

tional Table 1.

R E S U L T S

Description of studies

See: Characteristics of included studies, Characteristics of excluded

studies.

Results of the search

Searches were carried out in July 2010, and again in July 2012,

and August 2013, yielding 18,368 records of potential relevance

after de-duplication (July 2010: 11,822 records, July 2012: 4171

records, August 2013: 2375 records). Assessment of titles and ab-

stracts and elimination of duplicates between the searches resulted

in a list of 99 records for closer examination (Figure 1). One of

these articles is only available in French and is currently awaiting

classification pending translation (Baghdadli 2010).



Figure 1. Study flow diagram



Included studies

We included 22 studies involving 695 participants in this review;

in each case the main study is reported in a published journal article

and the dates of publication span from 1996 to 2013. There were

17 studies reported in a single published journal article: Solomon

2004; Fisher 2005; Golan 2006; Golan 2010; Kasari 2010; Landa

2011; Ryan 2010; Wong 2010; Begeer 2011; Hopkins 2011;

Kaale 2012; Williams 2012; Young 2012; Baghdadli 2013; Goods

2013; Schertz 2013; Wong 2013. In the case of Golan 2006, two

studies are reported of which only Experiment One is an RCT,

and therefore only this first data set is included in the review.

In addition, there are five studies for which data have been reported

in multiple outputs. These are a therapist-led theory of mind in-

tervention (Hadwin 1996), a computerised emotion recognition

intervention study (Bolte 2002), an imitation intervention study

(Ingersoll 2012), a music therapy study (Kim 2009), and a joint

attention and symbolic play intervention conducted by Kasari and

colleagues (Kasari 2006). In the case of the Kasari study, one out-

put is an unpublished PhD thesis (Arora 2008).

All 22 studies described themselves as randomised controlled tri-

als, and they were conducted in a wide variety of locations: Scandi-

navia (Bolte 2002; Kaale 2012); mainland Europe (Begeer 2011;

Baghdadli 2013); the UK and Ireland (Hadwin 1996; Fisher 2005;

Golan 2006; Golan 2010; Ryan 2010); the Far East (Kim 2009;

Wong 2010); Australia (Williams 2012; Young 2012); and the

USA (Solomon 2004; Kasari 2006; Kasari 2010; Landa 2011;

Hopkins 2011; Ingersoll 2012; Goods 2013; Schertz 2013; Wong

2013).

Participant baseline characteristics

Participants varied widely in age-range from preschoolers (e.g.

Kasari 2006) to adolescents and adults (e.g. Bolte 2002) but a ma-

jority focused on either pre-school or primary-school aged chil-

dren (see Characteristics of included studies). Almost all studies in-

cluded both boys and girls, though the proportion of male partici-

pants was much higher than females, corresponding to the known

greater prevalence of diagnosed ASD in males (Kogan 2009). Four

studies reported an all-male sample (Bolte 2002; Solomon 2004;

Kim 2009; Baghdadli 2013).

For all studies, a diagnosis of an ASD was a requirement for in-

clusion. A large proportion confirmed diagnosis using a clinical

instrument such as the Autism Diagnostic Observation Sched-

ule (ADOS, Lord 1994) or the Childhood Autism Rating Scale

(CARS, Schopler 1986). Two studies accepted prior clinical di-

agnosis as adequate (Hadwin 1996; Fisher 2005), but these also

instituted a checklist confirming that all diagnostic criteria were

met. Participants were reported as having a range of ASD diag-

noses, including autism, autism spectrum disorder, pervasive de-

velopmental disorder - not otherwise specified (PDD-NOS), high-

functioning autism (HFA), and Asperger’s syndrome (AS). Studies

recruiting participants with HFA and/or AS had participants in the

adolescent and adult age-range (e.g. Bolte 2002; Golan 2006) or

late childhood (Solomon 2004; Begeer 2011). Studies with young

children and preschoolers largely described participants as having

’core’ autism, or ASD.

All studies reported some measure of general intellectual ability

such as verbal mental age. Almost half of the included studies

included a sample in the normal intellectual range (Bolte 2002;

Solomon 2004; Golan 2006; Kim 2009; Golan 2010; Ryan 2010;

Begeer 2011; Young 2012; Baghdadli 2013) and the rest reported

on a sample with intellectual disability. One study split the par-

ticipant group into those with and without associated intellectual

delay (Hopkins 2011).

Sample sizes varied widely from n = 10 (Bolte 2002; Kim 2009)

to n = 61 (Kaale 2012). On the whole, very small proportions of

participants failed to complete the interventions. The maximum

drop-out rate was 27% from a small sample (Goods 2013), but

many studies reported no drop-out at all.

Intervention target types

The reported intervention types can be assigned to the following

categories, taken from the review protocol.

1. Interventions that explicitly state that they are designed to

teach ToM = (Hadwin 1996; Solomon 2004; Fisher 2005;

Begeer 2011; Baghdadli 2013).


teach precursor skills of ToM = (Bolte 2002; Golan 2006; Kasari

2006; Kim 2009; Golan 2010; Kasari 2010; Landa 2011; Ryan

2010; Wong 2010; Hopkins 2011; Ingersoll 2012; Kaale 2012;

Williams 2012; Young 2012; Goods 2013; Schertz 2013; Wong

2013).

3. Interventions that explicitly state that they are based on or

inspired by ToM models of autism.


test the ToM model of autism.

There were no studies falling into category three or four and the

vast majority of studies stated that they were designed to teach

precursor skills of ToM. Within this category we could also identify

some common intervention targets including the following.

• Emotion recognition (Bolte 2002; Golan 2006; Golan

2010; Ryan 2010; Hopkins 2011; Williams 2012; Young 2012)

• Joint attention and social communication (Kasari 2006;

Kim 2009; Kasari 2010; Landa 2011; Wong 2010; Kaale 2012;

Goods 2013; Schertz 2013; Wong 2013)

• Imitation skills (Ingersoll 2012)



Delivery methods, durations and dose

Three studies reported on the use of a computer program to de-

liver the intervention (Bolte 2002; Golan 2006; Hopkins 2011)

and all of these studies had emotion recognition as the target

skill. Three studies investigated the effect of a set of specially-de-

signed cartoons on emotion recognition (Golan 2010; Williams

2012; Young 2012). Other studies investigated the effects of one-

to-one therapist-led interventions (Hadwin 1996; Fisher 2005;

Kasari 2006; Landa 2011; Ryan 2010; Wong 2010; Ingersoll 2012;

Goods 2013) and two of these used the same manualised treatment

program (Kasari 2006; Kaale 2012). Some used a therapist-led ap-

proach in a group treatment setting (Solomon 2004; Begeer 2011;

Baghdadli 2013) and one was a group music therapy approach

(Kim 2009). Non-expert intervention delivery was rare with only

four studies reporting a parent-training element (Solomon 2004;

Kasari 2010; Begeer 2011; Schertz 2013) and one study report-

ing on teacher-training for intervention delivery in the classroom

(Wong 2013).

Intervention durations varied widely from two or three weeks (

Hadwin 1996; Young 2012) to six months (Landa 2011). Dose was

more consistent, with most falling within a range of 30 minutes

per week (Kim 2009) to 3.5 hours per week (Hadwin 1996; Kasari

2006; Golan 2010), and one outlying intervention which reported

therapist contact time of 2.5 hours per day (Landa 2011).

Most studies had wait-list or treatment-as-usual control con-

ditions. Six studies (Kim 2009; Landa 2011; Hopkins 2011;

Williams 2012; Young 2012; Baghdadli 2013) included control

conditions, which were not expected to have an impact on in-

tervention outcome but were included as a contact control only.

These included toy play, non-synchronous one-to-one time, using

art software, group leisure activities, and watching a Thomas the

Tank Engine DVD.

Outcome measures

On the whole, studies rarely identified a single primary outcome

measure. Those that organised outcomes into primary and sec-

ondary categories usually had multiple measures in each category.

The outcome measures used most commonly included the follow-

ing.

• Recognition of emotion from a variety of stimuli, including

static images of faces, static images of the eyes, film clips, short

stories, and cartoons (Bolte 2002; Solomon 2004; Golan 2006;

Golan 2010; Ryan 2010; Hopkins 2011; Williams 2012; Young

2012; Baghdadli 2013)

• Joint attention and joint engagement behaviours, often

measured using video coding of parent-child or teacher-child

interactions (Kasari 2006; Kim 2009; Kasari 2010; Landa 2011;

Ingersoll 2012; Kaale 2012; Goods 2013; Schertz 2013; Wong

2013)

• Direct assessment of ToM abilities (Hadwin 1996;

Solomon 2004; Fisher 2005; Begeer 2011; Williams 2012)

• Imitation skills (Landa 2011; Ingersoll 2012)

• Diagnostic outcome (Wong 2010, Young 2012)

The studies below included the following additional outcome mea-

sures.

• Caregiver measures such as quality of involvement,

adherence to treatment, mental health or satisfaction surveys

(Solomon 2004; Kasari 2010; Landa 2011, Wong 2010;

Baghdadli 2013; Wong 2013)

• General social skills measures, including rating scales and

observation (Kim 2009; Begeer 2011; Hopkins 2011; Ingersoll

2012; Williams 2012)

• Symbolic play measures (Hadwin 1996; Wong 2010) or

assessments of play variety (Goods 2013; Wong 2013)

• Language (Kasari 2006; Landa 2011) and conversational

skills (Hadwin 1996)

• fMRI (functional magnetic resonance imaging - assessment

of brain activity in facial recognition areas) (Bolte 2002)

• Adaptive function (Kasari 2006; Schertz 2013) and general

intellectual abilities (Landa 2011; Schertz 2013)

Selection for meta-analyses

Using protocol criteria, three groups of studies were identified as

eligible for meta-analysis.

1. Emotion recognition studies, with a treatment-as-usual

control, and outcome measures using judgements of emotional

expressions from static photographs of faces (Analysis 1.2).

2. Joint attention and social communication studies, with a

treatment-as-usual control, and outcome measures using coding

of parent-child interaction videos (Analysis 1.1).

3. Joint attention studies, with a treatment-as-usual control,

and outcome measures of joint attention initiation frequency

within a standardised assessment (the Early Social

Communication Scales) (Analysis 1.3).

Excluded studies

Examination of the abstracts and, where necessary, full texts of

reports resulted in a number of exclusions, listed in Characteristics

of excluded studies for the following reasons.

• Not fitting the ToM-linked criteria for inclusion (23

reports)

• Not presenting any new data (10 reports)

• Not randomised or quasi-randomised controlled trials (18

reports)

• Not reporting on a sample of people with ASD (one report)

• Reporting on a broad-based intervention without a specific

ToM-linked focus (12 reports)

• Diagnosis of participants invalid (one report)

• Reporting on an experimental pilot RCT with a very short

intervention period (three reports)



Risk of bias in included studies

Further information was requested from the majority of authors as

papers were not always complete in their reporting. The summaries

of ’Risk of bias’ judgements are shown in Figure 2 and Figure 3.

Figure 2. ’Risk of bias’ graph: review authors’ judgements about each risk of bias item presented as

percentages across all included studies.



Figure 3. ’Risk of bias’ summary: review authors’ judgements about each risk of bias item for each included

study.



Allocation

All studies in this review described themselves as randomised con-

trolled trials and were included on this basis. One was later revealed

to have used a non-random allocation procedure (Young 2012).

This study states that participants were “randomly allocated to twogroups” (Young 2012, p. 986) but email correspondence to clarify

the exact allocation procedure revealed that in fact this study used

alternate allocation by study enrolment. In other studies, a similar

statement is made but rarely is full detail provided.

Thus, only half of the included studies (11 reports) were judged to

have ’low’ risk of bias in terms of the description of the method of

randomisation. Only six were judged to have adequately described

allocation concealment. Therefore, the majority of included stud-

ies have either ’unclear’ or ’high’ risk of bias in this category.

In some cases, efforts were made to conceal allocation, for example,

using randomisation within blocks to ensure random allocation

and smooth delivery of the intervention (Goods 2013). However,

the use of blocks of fixed length meant that the final case within

each block would be allocated to a known condition.

Blinding

The majority of studies were judged at high risk of bias in this

category (19/22 studies, 86%). The three exceptions are Landa

2011 and Hopkins 2011 where partial blinding was achieved, and

Young 2012 who created a study design with full blinding as the

intervention was delivered not by a therapist but on a DVD.

Blinding of participants and personnel was rarely possible in the

studies included in this review, as behavioural interventions were

being used and these were often therapist-led. Blinding of outcome

assessors is easier to achieve and 14 studies (64%) clearly reported

blinding at this stage, though in a further five cases it was unclear

whether this was completed adequately.

Though risk of bias must be judged as high when blinding is not

achieved, a number of mitigating factors might help to reduce the

impact of this risk.

1. When working with very young children or those severely

affected by autism and/or intellectual disabilities, it is reasonable

to judge that participants are relatively oblivious to the

intervention content and certainly to the expected outcomes.

2. Likewise, although participants and parents may be aware

of their group they may not be apprised of the hypotheses of the

study. For example, Golan 2006 worked with able adults with

autism who were asked to “help in the evaluation of a piece of newsoftware” (p. 600) rather than being told the software was

designed to help them learn to understand emotions.

3. Many studies used automated outcome measures, especially

when using a computerised intervention (e.g. Bolte 2002; Golan

2006), which are more resilient to bias than experimenter-led

methods.

4. Studies using multiple outcome measures often achieved

blinding for a sub-set of those outcomes (e.g. Hopkins 2011).

Incomplete outcome data

There was very little evidence of attrition among the studies re-

ported here, and only three of the included studies were judged

to be at high risk of bias. The most extreme case of likely bias was

(Goods 2013) who reported 73% retention and analysed outcome

data for intervention completers only. Ten studies (45%) reported

outcome data for all of the original sample and where there was

participant drop-out this was usually described with clear reasons

to help the reader judge the impact of this drop-out. Where studies

were judged at unclear risk of bias, this was due to either a lack

of sufficient detail in the published report (e.g. Begeer 2011) or

because it was difficult to evaluate the impact of the drop-out on

findings (e.g. Kim 2009; Kasari 2006).

Selective reporting

Selective reporting was not evident among the papers included and

18 studies (82%) were judged at low risk of bias in this category.

However, it must be noted that the tendency not to identify a

primary outcome measure and to use multiple outcomes does

hinder conclusions about intervention efficacy.

One study (Bolte 2002) did not report means for a relevant out-

come measure - the International Affective Pictures System or

IAPS - for which there was no significant group difference. The

study authors were contacted to provide mean scores but were

unable to provide these due to the time elapsed since the study

and records not having been kept. One further study (Ryan 2010)

reported data in graphical form only, but the authors kindly pro-

vided accurate means and SDs for these data. Conversely, Goods

2013 reported non-significant findings in a table of results but

these were not discussed in the text.

Other potential sources of bias

One study reported a significant difference in ratings provided

by mothers and ratings provided by independent examiners (Kim

2009) with professional ratings providing a more positive estimate

of intervention efficacy. This bias is judged to be of low risk for

two reasons. First, the professional ratings used to construct the

primary outcome for the intervention were blind to group. Sec-

ond, the authors provided a reasonable justification for the under-

estimation of intervention efficacy by parent ratings, which is that

mothers over-estimate pre-intervention abilities of their children,

and thus under-estimate efficacy of the intervention.



Another study combined the wait-list control group with the inter-

vention group to provide a larger sample size for analysis of some

intervention efficacy measures (Wong 2010). However, prior to

this stage in the analysis, between group comparisons were also

made and these provided the primary outcomes for the study. The

impact of this bias is judged to be unclear as while a between group

comparison was made, the long-term maintenance of intervention

gains is disguised by the combination of data sets at the final time

point.

Schertz 2013 adopted a variable intervention period which could

have weighted findings towards a positive conclusion regarding in-

tervention efficacy. Their strategy was to recruit participants who

were demonstrably lacking the target joint attention skills. Partic-

ipants were then paired and randomly assigned to intervention or

treatment-as-usual control groups. Intervention then proceeded

until an individual participating child had achieved the target skill.

At this point, exit assessment measures were taken for that child

and their matched pair. Therefore, within each pair, participants

experienced the same interval between baseline and post-interven-

tion assessment. However, when analysed as a group, this system

ensured that every child in the intervention group had shown sig-

nificant gains in the target skill, thus biasing the study towards a

positive conclusion.

No other potential sources of bias were identified in the studies

selected for inclusion in this review. Also, the authors note some

examples of particularly good practice in the prevention of bias,

including close measurement of treatment adherence in therapist-

led (Baghdadli 2013) and parent-training studies (Schertz 2013).

Effects of interventions

See: Summary of findings for the main comparison

Studies in this review used a wide variety of outcome measures,

often using measures specific to their intervention target and

sometimes designed specifically for that study. In addition, effect

sizes, mean differences reported as standardised mean differences

(SMD) and confidence intervals (CI) were not always reported,

though we include these below where available. Intervention mea-

sures are listed in additional Table 2 and discussed below, organ-

ised by primary and secondary outcome category. In addition, pri-

mary outcome results are collated in the Summary of findings for

the main comparison.

Evaluating primary outcomes: communication

Primary outcome measures in this section were those using stan-

dardised assessments to assess communication skills of diagnostic

relevance (i.e. more than just expressive language).

Two studies employed diagnostic assessment measures to evalu-

ate change in symptom level within the communication domain.

Wong 2010 used a sub-set of ADOS (Lord 2000) items to eval-

uate communication gains in response to intervention, finding

improvements in relevant items (vocalisation directed to others,

gestures, pointing) in the intervention group (median difference

= 4 points), but not in the control group (median difference = 2.5

points). This finding is weakened by the fact that these analyses

compared change from baseline to outcome in each group sepa-

rately and there was no between-group comparison. Furthermore,

the ADOS is not intended as an intervention outcome measure,

and it is not usual to analyse a sub-set of items. On the other

hand, this finding is strengthened by a comparison that shows no

intervention group gains in items pre-identified as non-relevant to

the intervention. Young 2012 similarly reported change for indi-

vidual items of the Social Communication Questionnaire (SCQ)

(Rutter 2003). Of specific relevance in this outcome category, they

analysed change in eye contact and gaze aversion, and found no

intervention effects on these items (effect sizes: ηp2 = 0.001 and

ηp2 = 0.002 respectively).

For participants at a higher level of communicative sophistication,

Hadwin 1996 (reported in Hadwin 1997) evaluated the impact

of ToM intervention on complex language skills. They found no

effect of intervention on conversational skills, and also raised ad-

ditional evidence that language level may moderate intervention

effects when teaching ToM skills (Hadwin 1996) though this is

not explored elsewhere.

Evaluating primary outcomes: social function

Primary outcome measures in this section were those using stan-

dardised assessments to assess social skills of diagnostic relevance.

Six studies used the Early Social Communication Scales (ESCS)

to evaluate the outcome, standardised observational assessment of

social engagement behaviours, including joint attention (Kasari

2006; Kim 2009; Ingersoll 2012 ;Kaale 2012; Goods 2013; Wong

2013). Due to differences in measurement (e.g. reporting be-

haviour frequency versus amount of time; reporting sub-items

from the scales versus reporting scale totals) and study design (those

with or without a therapeutic control condition), not all studies

could be combined for meta-analysis. However, three studies were

combined in this way (Analysis 1.3) and the outcome indicated no

significant intervention effect on social behaviour measured in this

way (SMD 0.23, 95% CI -0.48 to 0.94, Z = 0.63, P value = 0.53,

three studies, 92 participants). There was no evidence of hetero-

geneity in effects (I2 = 57%, Chi2 (df = 2) = 4.66, P value = 0.10,

Tau2 = 0.22) (Figure 4). Among studies not included in meta-

analysis, Kasari 2006 found large treatment gains in the showing

(effect size = 1.50) and responding to joint attention (effect size =

1.20) items of the ESCS but not in other relevant items such as

pointing and giving. This may indicate a lack of generalisation of

skills beyond the specific taught items.



Figure 4. Forest plot of comparison: 1 test, outcome: 1.3 Joint attention initiations in standardised

assessment.

As described above, two studies used scores on a diagnostic or

screening measure as their primary outcome, principally focusing

on change in social function. Wong 2010 reported item-by-item

changes rather than algorithm totals for the ADOS (Lord 2000),

and Young 2012 analysed intervention effects on a single ’Social

Peer Interest’ item of the SCQ (Rutter 2003). Young 2012 did

not find significant effects on this SCQ item (ηp2 = 0.06), despite

the intervention having a positive impact on emotion recognition

skill; evidence of a lack of generalisation or expansion of a taught

skill to related domains. In contrast, Wong 2010 did report inter-

vention effects on items from the ADOS, which are relevant to

the intervention content (intervention group median difference

= 7 points; control group median difference = 2.5 points), and

crucially not for items unrelated to the intervention. However, it

should once again be noted in both studies that the measures used

were not designed to be analysed on an item-by-item basis. More-

over, Wong 2010 reported no direct comparison of the degree of

change between the two groups.

Williams 2012 additionally reported on social abilities measured

in a real-world context using standardised measures of life skills

(intervention group mean difference = 2.13 points; control group

mean difference = 1.59 points). No evidence was found of in-

tervention effects measured in this way, once more indicating a

lack of generalisation from specifically-taught skills to wider social

abilities.

Further detail on outcomes from observed measures of social en-

gagement is provided below under the heading of Secondary Out-comes: Change in participant behaviour or quality of interpersonalinteraction, or both, measured by direct observation.

Evaluating secondary outcomes

Intervention Specific: Change in targeted cognitive skill,

Emotion recognition

Seven studies examined the impact of intervention on facial affect

recognition skills from photographs, as compared to treatment-as-

usual (Solomon 2004; Golan 2006; Ryan 2010; Hopkins 2011;

Williams 2012; Young 2012; Baghdadli 2013), which were in-

spected further for potential inclusion in a meta-analysis. Young

2012 was excluded from the meta-analysis due to use of a non-

random allocation procedure. Baghdadli 2013 was eligible for in-

clusion in a meta-analysis in principle, but it was not possible to

extract the relevant data from the paper, which reported median

scores and inter-quartile range for it’s non-normally distributed

data, rather than means and standard deviations. The study found

a significant improvement in the intervention group (but not the

control group) in recognition of angry emotions only (effect size,

Cohen’s d = -0.8, P value = 0.05), but no significant differences

between groups for other emotions. It was also inappropriate to

incorporate Williams 2012 into the meta-analysis as this study

used a control condition, which was hypothesised to have poten-

tial treatment effects.

Hopkins 2011 reported on two separate samples: children with

low-functioning autism (LFA) and children with high-functioning

autism (HFA). All other studies in this group reported on partici-

pant samples with IQ or language ability in the normal range, and

therefore it seemed most appropriate to include the HFA sample

from Hopkins 2011 in the meta-analysis.

Likewise, Solomon 2004 reported separately on younger and older

groups of children. The majority of other studies in this group

reported on child participants whose age more closely matches

the older group of Solomon et al (Ryan 2010; Hopkins 2011),

and one remaining study involved an adult sample (Golan 2006).

Therefore, it was decided to include the older participant group

in the meta-analysis.

All studies in the emotion recognition group reported significant

group differences on outcome immediately post-treatment, mea-

sured by recognition of facial emotion from static images. The

meta-analysis (Analysis 1.2) shows evidence of a positive interven-

tion effect on emotion recognition (SMD 0.75, 95% CI 0.22 to

1.29, Z = 2.75, P value = 0.006, four studies, 105 participants).

There was no evidence of heterogeneity in effects (I2 = 36%, Chi2 (df = 3) = 4.70, P value = 0.19, Tau2 = 0.11) (Figure 5). A study

excluded for reasons of bias (Young 2012) also found a positive

effect of intervention on emotion recognition skills.



Figure 5. Forest plot of comparison: 1 test, outcome: 1.2 Emotion recognition from face photographs, TAU

control.

We note that Bolte 2002 reported a null finding in a related

study using emotion recognition training, with recognition of fa-

cial emotion from static images as an outcome. However, it was

not possible to retrieve these data. This study had a small sample

size (n = five per group) and therefore it is unlikely that this null

finding would have a large effect on the reported meta-analysis.

In addition to the results included in our meta-analyses, many

interventions targeting emotion recognition skills were also evalu-

ated by additional measures tapping the same or related constructs.

For example, emotion recognition was assessed using stimuli, in-

cluding audio clips (Golan 2006), film clips (Golan 2006), emo-

tional vocabulary tests (Golan 2010), and matching emotional vi-

gnettes to facial expressions (Golan 2010). A consistent finding

in these studies using multiple measures of the same construct

was an absence of generalisation of the target skill to novel set-

tings or stimuli where this was assessed. For example, Golan 2006

evaluated emotion recognition and found improvements only on

’close-generalisation’ tasks, which did not extend to other outcome

measures (e.g. reading-the-mind-in-the-eyes: intervention group

mean difference = + 0.7 points; control group mean difference =

-0.9 points).


Theory of Mind (ToM)

Four studies, all aiming to directly teach ToM, used explicit assess-

ment of ToM as their primary outcome (Hadwin 1996; Solomon

2004; Fisher 2005; Begeer 2011). These outcome measures over-

lap with those described in the Emotion Recognition section above

as they sometimes use emotional content (especially complex emo-

tions and mental states). However, they are evaluated indepen-

dently here as the source authors clearly identify them as assess-

ments of ToM. Once again, positive outcomes were found when

assessing progress within, or close to, the taught context, but there

was an absence of generalisation of taught skills to novel, or more

complex scenarios or to abilities hypothetically built on ToM. For

example, Begeer 2011 reported a significant difference between

groups in degree of improvement measured by the ToM test (ef-

fect size, Cohen’s d = 0.75), but no such effect in measures of self-

reported empathy or parent-reported social skills.

Williams 2012 reported on an emotion skills intervention but

additionally assessed ToM abilities in order to explore the extent

of skills learnt. Once more, there were no intervention effects on

this extended skill set.


Imitation

Two studies reported gains in imitation skills as their primary out-

come in intervention studies with toddlers (Landa 2011; Ingersoll

2012). For example, Ingersoll 2012 (reported in Ingersoll 2010)

showed that the treatment group made larger gains in imitation

than the control group, though this finding had a small to mod-

erate effect size (elicited imitation, ηp2 = 0.20; spontaneous imi-

tation, ηp2 = 0.29; object imitation ηp

2 = 0.21; gesture imitation

ηp2 = 0.38). Unfortunately, due to differences in measurement it

was not possible to combine these two studies in meta-analysis.

Their combined sample size is just 69 participants.

Intervention Specific: Change in targeted cognitive skill, Play

Finally, four studies included assessment of play as a secondary

outcome (Hadwin 1996, Wong 2010; Goods 2013; Wong 2013).

Hadwin 1996 found no effect of teaching ToM understanding on

observed symbolic play skills, while the findings of Wong 2010,

using an observational symbolic play test are positive, though the

larger sample size is reported by Hadwin 1996. Regarding the

range of play types observed, Goods 2013 reported positive in-

tervention effects on this variable using a ’Structured Play Assess-

ment’ while Wong 2013 reported no positive effects on the same

measure. These two studies each reported a different output from

the ’Structured Play Assessment’ (play types versus play level) mak-

ing it illogical to combine these data in a meta-analysis.

Change in participant behaviour or quality of interpersonal

interaction, or both, measured by direct observation



A series of studies used joint engagement behaviours during adult-

child interactions as an outcome measure. However, subtle but

important differences in the operationalisation of these outcomes

meant that very few were eligible for combined analysis. For ex-

ample, some studies reported on parent-child (Kasari 2006; Kasari

2010) and some on teacher-child interactions (Kim 2009; Kaale

2012; Goods 2013). Some studies reported on only one category

of joint attention behaviour as a single outcome (Kaale 2012),

while some sub-divided these into initiations and responses (Kasari

2006; Kasari 2010; Landa 2011). In addition, there were dif-

ferences in the outcome unit of measurement with Kasari 2006

reporting total seconds of joint attention behaviours and Kasari

2010 reporting frequency of joint attention behaviours. And fi-

nally, some studies reported on behaviour measured in a one-to-

one setting (Kaale 2012) while others reported on spontaneous

behaviour within a group setting, e.g. the classroom (Wong 2013)

or during school recess (Hopkins 2011).

The only outcomes of sufficient similarity to be appropriate for

inclusion in meta-analysis were the measures of joint engagement

during mother-child play reported in Kasari 2010 and Kaale 2012,

both as percentages of total time. In meta-analysis (Analysis 1.1)

there was evidence of a positive intervention effect on joint en-

gagement (SMD 0.55, 95% CI 0.11 to 0.99, Z = 2.45, P value

= 0.01, two studies, 88 participants). There was no evidence of

heterogeneity in effects (I2 = 5%, Chi2 (df = 1) = 1.05, P value =

0.30, Tau2 = 0.01) (Figure 6).

Figure 6. Forest plot of comparison: 1 test, outcome: 1.1 Joint engagement in mother-child interaction.

Here, there was a little evidence that taught skills may generalise

to new settings. Kasari 2006 and Kaale 2012 both found that

therapist-taught social communication skills were in evidence in a

parent-child interaction scenario and when using novel assessment

materials (Gulsrud 2007). For example, Kaale 2012 found that

children in the intervention group spent on average 12.2% more

time in a joint engagement state with their mother compared with

children in the control group (95% CI = 2.4% to 22%, effect size

Cohen’s d = 0.67).

Change in participant behaviour or skills measured by adult

report

Other studies reported an array of general social skills measures

(Solomon 2004; Fisher 2005; Kim 2009; Wong 2010; Begeer

2011, Hopkins 2011; Ingersoll 2012). These were very different in

method of assessment, construct being examined, and data format,

and so it is not possible to make a direct comparison between

studies. Nevertheless, the overall message from the study authors

is of improvement in social skills as a result of intervention.

Change in participant cognitive skill, measured by

standardised assessment

Three studies reported measures of language and general cognitive

or adaptive ability as an outcome (Kasari 2006 reported in Kasari

2008, Landa 2011; Schertz 2013). The studies produced conflict-

ing results. Landa 2011 did not find a significant difference be-

tween intervention and control groups on a measure of expressive

language despite a moderate effect size of 0.49, while Kasari 2008

and Schertz 2013 reported significant gains in expressive language

in the intervention compared with the control group. However,

when evaluating participants over the long term (Kasari 2012a),

there was no evidence that treatment continued to impact on lan-

guage and cognitive outcome five years from baseline.

Acceptability of Intervention

A range of studies incorporated caregiver measures such as quality

of involvement, adherence to treatment, mental health or partic-

ipant well-being and satisfaction surveys (Solomon 2004; Kasari

2010; Wong 2010; Baghdadli 2013; Wong 2013), but these were

very different in method of assessment and specific construct tar-

geted, and so it is not possible to draw solid conclusions from these

data.

Rate of drop-out



As discussed previously (Assessment of risk of bias in included

studies), rates of drop-out in the included studies were very low

with half of the included studies reporting that the full sample

remained enrolled for the length of the trial. One study reported

a very high rate of drop-out from a small sample in a study taking

place within a pre-school setting and it was unclear why this drop-

out rate was so high (Goods 2013). Otherwise, the highest rates

of drop-out occurred when the intervention was self-directed by

an individual with ASD (Golan 2006) or when the intervention

took place at a clinic which required significant travelling time for

participants (Kim 2009).

Economic data

No studies reported economic data.

Follow-up effects

A minority of studies followed up participants after the immedi-

ate intervention period had ended (Hadwin 1996; Fisher 2005;

Kasari 2006; Kasari 2010; Landa 2011; Ryan 2010). Follow-up

periods ranged from six weeks to five years and findings consis-

tently showed maintenance of treatment gains in a range of mea-

sured skills, including ToM (Hadwin 1996; Fisher 2005), social

engagement (Kasari 2006; Kasari 2010; Landa 2011), and emo-

tion recognition (Ryan 2010). In some cases there was evidence of

continued significant growth during the post-intervention period

(Fisher 2005; Kasari 2006; Kasari 2010). The sole exception is

Kasari 2006 (reported in Kasari 2012a) who found no long-term

impact of intervention on language and cognitive outcome at five

years post-baseline.

D I S C U S S I O N

Summary of main results

Twenty-two randomised trials were included in this review. All

reported on interventions that targeted either Theory of Mind

(ToM), or one of the accepted sub-components of ToM such as

shared attention, emotion recognition or imitation. Most involved

either wait-list or treatment-as-usual control conditions, or a con-

trol condition devised to replicate contact time but without ther-

apeutic content.

Risk of bias in the included studies was variable. Very few stud-

ies provided adequate information and in a majority of cases the

authors were contacted to provide further details. In particular,

details of sequence generation and allocation concealment were

lacking in the published articles. Blinding of participants and per-

sonnel was judged to be impossible in almost every study reviewed,

and as a result risk of bias in this category was high. Blinding of

outcome assessors is better achieved and reported on, and concerns

about selective reporting or attrition were rare.

In addition, outcomes varied widely, both in the construct being

measured and the means of measurement. This made it a challenge

to combine studies for meta-analysis and as a result only three,

very small meta-analyses were conducted. Outcomes also differed

significantly from those predicted in the review protocol. There

were very few outcomes reported that fell into the protocol-de-

fined categories of symptom level for the two diagnostic domains

of social and communication impairments. Instead, studies mostly

reported on intervention-specific measures of constructs such as

emotion recognition, joint attention and social communication,

and imitation. In these three intervention target areas (emotion

recognition, joint attention and social communication, and imi-

tation), there was modest evidence of intervention success.

Emotion recognition

Studies in the emotion recognition field consistently found pos-

itive effects of specific training, a conclusion reinforced by com-

bined analysis of four studies (Figure 5). However these skills did

not always generalise to testing contexts that moved beyond the

trained content. Since the overall goal of emotion recognition

training for people with ASD must be to improve real world emo-

tion recognition and, by extension, emotional understanding, this

is a significant limitation, which calls into question the value of

emotion recognition interventions. These studies are additionally

limited by their reliance on both teaching and measuring facial

affect recognition as a proxy for emotion recognition. Real world

emotion recognition skills require appraisal of emotional valence

from a variety of sources (e.g. facial emotion, body language, con-

tent and tone of speech, context) and these skills are rarely taught.

Joint attention and social communication

A number of included studies reported on therapist-led, one-to-

one or group interventions, normally with young children with

autism, targeting a core deficit of joint attention and social com-

munication. These studies often involved a high level of inter-

vention contact hours but could produce sustained effects (Kasari

2010), although the intervention effect may not be maintained

in the longer term, i.e. over a period of years (Kasari 2006). The

studies in this category have significant real world relevance as

there is evidence that therapist training can lead to improvements

in interactions with other familiar adults as well (Kasari 2006;

Kaale 2012). Since joint attention is a theoretical prerequisite for

both language learning and ToM development, more longitudinal

studies looking at the effect of joint attention improvement on

these linked skills would be of value.

Imitation



One study specifically targeted imitation both as intervention tar-

get and method of delivery (Ingersoll 2012), while another used

a more general social communicative approach which impacted

on imitation skills (Landa 2011). These studies, in common with

those described above, require further investigation to explore

whether training on this theoretical building block of ToM can

subsequently enhance ToM development.

Theory of Mind

Perhaps surprisingly, a minority of included studies focused ex-

plicitly on training ToM skills. As for emotion recognition, it was

found relatively straightforward to teach a core ToM skill and to

find a difference in that skill when tested under the same circum-

stances, but generalisation across props, settings, and partners was

a greater challenge.

General conclusions

A common theme, therefore, in these studies was the difficulty

of creating positive intervention effects recognisable in everyday

function. The skills targeted in the interventions included in this

review are all developmentally linked, both to each other and to

more general abilities such as language and friendship skills. How-

ever, it is rare for an intervention to succeed in triggering this

developmental chain. For this reason it may be that the broad-

based interventions, excluded here because they cannot be clearly

categorised according to the protocol criteria, represent a more

successful approach to intervention for autism. They may help to

promote generalisation by working in a more ecologically valid

way to support the development of new skills in combination.

One complicating factor, which hinders the opportunity to draw

strong conclusions from the review, is that the studies lack ap-

propriate outcome measures in two different ways. First, there is

no consistent, widely-used measure for assessing intervention out-

come using diagnosis-relevant domains for ASD research - not

even for a specific age-range or ability level. As a result, studies

often rely on bespoke measures such as video-coding of parent-

child interactions, or multiple measures assessing a range of social,

intellectual, and adaptive skills. This is an obstacle to comparison

of multiple studies both at a systematic review level and for the av-

erage reader of these published reports. Second, the use of multiple

measures, combined with an absence of explicit identification of a

single primary outcome measure, means that it is often possible to

claim intervention success on a sub-set of the measures used, but

not all. Without a priori identification of the primary outcome

measure it is difficult to judge how these mixed findings should

be interpreted.

Overall completeness and applicability ofevidence

The studies reported in this review cover a good range of ToM

and associated skills, though the fact of this array means that more

evidence is needed in each category. It was not possible to con-

duct subgroup analyses for this review, but it is theoretically likely

that we would find differences between specific diagnostic cate-

gories, age groups, and intervention delivery methods. The evi-

dence, though limited and of generally low quality, suggests that

it may be possible to teach ToM-linked abilities.

Research now should explore how different approaches might be

applied to different populations, requiring a much larger and more

systematic body of evidence. In particular, it is understood that

different age and ability levels require support developing different

sub-skills and mapping out these relationships would be of value.

In the field of emotion recognition, however, this skill has been

successfully taught to preschoolers, children and adults, quite often

using computer-based methods, and an understanding of why this

is a difficulty which persists across the life span would be of value.

Quality of the evidence

The evidence included here varies in quality due to the aforemen-

tioned difficulty of blinding participants and personnel, and un-

der-reporting of other relevant practices such as sequence gener-

ation and allocation concealment. In addition, sample sizes are

often very low, with the maximum reported at only n = 61 (Kaale

2012). Due to a lack of strong reporting norms it has not always

been possible to accurately judge risk of bias in the studies re-

ported here, which may have unknown impact on conclusions.

Further, while attrition rates are low on the whole, all studies anal-

yse only the final sample and do not use an intention-to-treat

analysis. Once more it is impossible to judge to what degree this

may have affected results, and in what direction. Overall, there is

a lack of good quality evidence in this field and a requirement for

more randomised controlled trials representing highest standards

in methodology, particularly outcome assessment.

One group of studies represented a good example of independent

evaluation of the same intervention, The Transporters DVD for

teaching emotion recognition and understanding (Golan 2010;

Williams 2012; Young 2012). Unfortunately, minor but impor-

tant differences in study design between these reports meant that

they could not be combined in a meta-analysis. However this rare

example of replication, perhaps because the DVD-based interven-

tion is relatively easy to apply, is a notable and positive exception

in a field of diverse intervention strategies.

We found no evidence of adverse effects but this partly reflects a

lack of attention to these in the source literature. There seems to

be a common presumption that behavioural interventions, such as

those reviewed here, do not carry a risk of harm but this possibility

needs to be addressed explicitly in future studies.



Potential biases in the review process

The review authors were restricted by only evaluating studies avail-

able in English language. In addition, some databases searched at

the first data collection point were no longer available to us when

searches were repeated two years later. It is unclear how these re-

strictions may have affected the conclusions drawn. Dissertation

databases were not searched independently and instead the review

authors requested unpublished data, including dissertations, from

key authors. Two relevant PhD theses were examined (Arora 2008;

Rodgers 2012) one of which is an excluded study (Rodgers 2012).

We note that it is extremely rare in this field for postgraduate dis-

sertations to conduct an independent clinical trial because of the

challenges surrounding recruitment and design.

Agreements and disagreements with otherstudies or reviews

As this review appears to be the first to consider ToM interven-

tions as a group of studies, no comparison can be made with the

conclusions of other reviews.

A U T H O R S ’ C O N C L U S I O N S

Implications for practice

The review suggests it may be possible to teach both Theory of

Mind (ToM) and the precursor skills associated with the construct.

However this teaching rarely or never generalises to novel contexts,

and it is unclear whether there is long-term maintenance of learnt

skills, or developmental progression in learning. Future reviewers

should consider investigating the results of multi-modal interven-

tions, which were excluded from this review, and which teach a

range of real-world social and communicative skills as well as aca-

demic learning. Given the problems with maintenance and gen-

eralisation of taught skills, multi-modal approaches may be more

successful in improving social and communication outcomes in

people with ASD.

Implications for research

The ToM model of autism development proposes that ToM is a

core deficit, which links both to precursor skills, such as joint at-

tention and emotion recognition, and to subsequent abilities such

as making friends and understanding metaphorical language. The-

oretically then, this developmental model implies that a successful

intervention at one point in the chain should have impact all the

way up the developmental ladder.

More longitudinal studies are required to establish whether teach-

ing a specific skill at one time point will lead not only to mainte-

nance of that skill but also the development of further associated

abilities subsequently. Studies systematically linking intervention

targets and delivery methods to participant age and ability would

support this process by helping to identify the right ’starting place’

for an intervention for a particular individual. In order to reach

larger populations, multi-site studies are required, with co-opera-

tion across research centres and national borders. This is challeng-

ing not just in a logistical sense but also because countries and even

counties vary in the service provision and intervention approaches

offered to people with ASD across ages.

There is an urgent need for better outcome measures for autism re-

search across the board. This has been recognised in current fund-

ing from the National Institute for Health Research Health Tech-

nology Assessment programme for a systematic review of measure-

ment properties of outcome measures for children with autism up

to the age of six years (http://www.hta.ac.uk/project/2830.asp).

For future studies in the field of ToM interventions, ideally out-

come measures should capture symptom severity in core diagnostic

domains, without looking for a change in diagnostic status - which

is neither feasible nor necessarily to be desired. In the short term,

however, the outcome measure issue could be partially resolved

by better study reporting, in particular a priori identification of

a single primary outcome measure to define intervention success.

Additionally, secondary measures should have a clear rationale for

inclusion and should be, wherever possible, linked to intervention

components. Improvements in reporting could also be made, es-

pecially in providing details of sequence generation and allocation

concealment, which is rarely described.

The Theory of Mind model of autism was first introduced in 1985

and in 2000 a review of the changes in the model was published

(Baron-Cohen 2000). Now, almost another 15 years on, multi-

ple versions (Astington 2011) and alternative explanatory models

abound (e.g. Happe 2006; Mottron 2006; Baron-Cohen 2010)

and there have been direct attacks both on the model (Hobson

1991) and on the methods used to measure the construct (Bloom

2000). One meaningful way to evaluate the explanatory power

and clinical and educational relevance of these competing theories

is to explore their impact in an intervention setting. Currently,

however, the quality and quantity of evidence needed even for

ToM alone is inadequate to do so.

A C K N O W L E D G E M E N T S

The authors are grateful for the support of Jo Abbott, Margaret An-

derson, Chris Champion, Jane Dennis, Laura MacDonald, Geral-

dine MacDonald, and Inalegwu Oono for their help in develop-

ing this review and for the comments of Michelle Dawson on a

previous draft.



R E F E R E N C E S

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high-functioning autism: a pilot randomized controlled

trial. European Child & Adolescent Psychiatry 2013;22(7):

433–42.

Begeer 2011 {published data only}

Begeer S, Gevers C, Clifford P, Verhoeve M, Kat K,

Hoddenbach E, et al.Theory of mind training in children

with autism: a randomized controlled trial. Journal of

Autism and Developmental Disorders 2011;41(8):997–1006.

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Dierks T, Poutska F. Facial affect recognition training in

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Fisher 2005 {published data only}

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and executive function in children with autistic spectrum

disorders. Journal of Autism and Developmental Disorders2005;35(6):757–71.

Golan 2006 {published data only}

Golan O, Baron-Cohen S. Systemizing empathy: teaching

adults with Asperger syndrome or high-functioning

autism to recognize complex emotions using interactive

multimedia. Development and Psychopathology 2006;18(2):

591–617.

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Golan O, Ashwin E, Granader Y, McClintock S, Day K,

Leggett V, et al.Enhancing emotion recognition in children

with autism spectrum conditions: an intervention using

animated vehicles with real emotional faces. Journal of


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autism: pilot RCT. Journal of Autism and DevelopmentalDisorders 2013;43(5):1050–6.

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teach children with autism to understand emotions, belief

or pretence?. Development and Psychopathology 1996;8(2):

345–65.

Hadwin J, Baron-Cohen S, Howlin P, Hill K. Does teaching

theory of mind have an effect on the ability to develop

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Hopkins 2011 {published data only}

Hopkins IM, Gower MW, Perez TA, Smith DS, Amthor

FR, Wimsatt FC, et al.Avatar assistant: improving social

skills in students with an ASD through a computer-based

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intervention of social functioning in children with autism.

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1768–73.

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trial of preschool-based joint attention intervention for

children with autism. Journal of Child Psychology and

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Kasari 2006 {published data only}

Arora T. Perseveration in young children with autism and the

association with joint attention [PhD Thesis]. University of

California, 2008.

Gulsrud AC, Kasari C, Freeman S, Paparella T. Brief report:

children with autism’s response to novel stimuli while

participating in interventions targeting joint attention or

symbolic play skills. Autism 2007;11(6):535–46.∗ Kasari C, Freeman S, Paparella T. Joint attention and

symbolic play in young children with autism: a randomized

controlled intervention study. Journal of Child Psychologyand Psychiatry 2006;47(6):611–20.

Kasari C, Gulsrud A, Freeman S, Paparella T, Hellemann

G. Longitudinal follow up of children with autism receiving

targeted interventions on joint attention and play. Journalof the American Academy of Child and Adolescent Psychiatry

2012;51(5):487–95.

Kasari C, Paparella T, Freeman S, Jahromi LB. Language

outcome in autism: a randomised comparison of joint

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Kasari 2010 {published data only}

Kasari C, Gulsrud A, Wong C, Kwon S, Locke J.

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of Autism and Developmental Disorders 2010;40(9):1045–56.

Kim 2009 {published data only}∗ Kim J, Wigram T, Gold C. Emotional, motivational

and interpersonal responsiveness of children with autism

in improvisational music therapy. Autism 2009;13(4):

389–409.

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music therapy on joint attention behaviours in autistic



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Landa 2011 {published data only}

Landa RJ, Holman KC, O’Neill AH, Stuart EA.

Intervention targeting development of socially synchronous

engagement in toddlers with autism spectrum disorder: a

randomized controlled trial. Journal of Child Psychiatry and

Psychology 2011;52(1):13–21.

Ryan 2010 {published and unpublished data}

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skills to children with autism. Journal of Autism and

Developmental Disorders 2010;40(12):1505–11.

Schertz 2013 {published data only}

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joint attention mediated learning for toddlers with autism

spectrum disorders: an ititial randomized controlled study.

Early Childhood Research Quarterly 2013;28(2):249–58.

Solomon 2004 {published data only}

Solomon M, Goodlin-Jones BL, Anders TF. A social

adjustment enhancement intervention for high functioning

autism, Asperger’s syndrome and pervasive developmental

disorder NOS. Journal of Autism and DevelopmentalDisorders 2004;34(6):649–68.

Williams 2012 {published data only}

Williams BT, Gray KM, Tonge BJ. Teaching emotion

recognition skills to young children with autism: a

randomized controlled trial of an emotion training

programme. Journal of Child Psychology and Psychiatry 2012;

53(12):1268–76.

Wong 2010 {published data only}

Wong VCN, Kwan QK. Ramdomized controlled trial for

early intervention for autism: a pilot study of the Autism 1-

2-3 Project. Journal of Autism and Developmental Disorders

2010;40(6):677–88.

Wong 2013 {published data only}

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teachers of young children with autism: a randomized

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Young 2012 {published data only}

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984–91.

References to studies excluded from this review

Adams 2012 {published data only}

Adams C, Lockton E, Freed J, Gaile J, Earl G, McBean

K, et al.The Social Communication Intervention Project:

a randomized controlled trial of the effectiveness of speech

and language therapy for school-age children who have

pragmatic and social communication problems with or

without autism spectrum disorder. International Journal ofLanguage & Communication Disorders 2012;47(3):233–44.

Aldred 2004 {published data only}

Aldred C, Green J, Adams C. A new social communication

intervention for children with autism: a pilot randomised

controlled treatment study suggesting effectiveness. Journal

of Child Psychology and Psychiatry 2004;45(8):1420–30.

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Beaumont 2008 {published data only}

Beaumont R, Sofronoff K. A multi-component social

skills intervention for children with Asperger syndrome:

the Junior Detective Training Program. Journal of Child

Psychology and Psychiatry 2008;49(7):743–53.

Cardon 2011 {published data only}

Cardon TA, Wilcox MJ. Promoting imitation in young

children with autism: a comparison of reciprocal imitation

training and video modelling. Journal of Autism and


Carter 2011 {published data only}

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AS, Yoder P. A randomized controlled trial of Hanen’s ’More

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Dawson 2010 {published data only}

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interventions to promote the generalization of social

competence for adolescents with high-functioning autism

and Asperger’s syndrome. Exceptionality 2012;20(2):

94–113.



Silver 2001 {published data only}

Silver M, Oakes P. Evaluation of a new computer

intervention to teach people with autism of Asperger

syndrome to recognise and predict emotions in others.

Autism 2001;5(3):299–316.

Smith 2000 {published data only}

Smith T, Buch GA, Gamby TE. Parent-directed, intensive

early intervention for children with pervasive developmental

disorder. Research in Developmental Disbailities 2000;21(4):

297–309.

Smith 2004 {published data only}

Smith T, Groen AD, Wynn JW. Randomized trial of

intensive early intervention for children with pervasive

developmental disorder. In: Feldman MA editor(s). EarlyIntervention: The Essential Readings. Blackwell, 2004:

153–81.

Stichter 2001 {published data only}

Stichter JP. Functional analysis: the use of analogues in

applied settings. Focus on Autism and Other Developmental

Disabilities 2001;16(4):232–9.

Strain 2011 {published data only}

Strain PS, Bovey EH. Randomized, controlled trial of the

LEAP model of early intervention for young children with

autism spectrum disorders. Topics in Early Childhood Special

Education 2011;31(3):133–54.

Swettenham 1996 {published data only}

Swettenham J. Can children with autism be taught to

understand false belief using computers?. Journal of Child

Psychology and Psychiatry 1996;37(2):157–65.

Tanaka 2010 {published data only}

Tanaka JW, Wolf JM, Klaiman C, Koenig K, Cockburn J,

Herlihy L, et al.Using computerized games to teach face

recognition skills to children with autism spectrum disorder:

the Let’s Face It! program. Journal of Child Psycholgy andPsychiatry 2010;51(8):944–52.

Temple 2007 {published data only}

Temple K. A randomized comparison of the effect of

two prelinguistic communication interventions on the

acquisition of spoken communication in preschoolers with

ASD. Child: Care, Health and Development 2007;33(3):

348–9.

Thomeer 2012 {published data only}

Thomeer ML, Lopata C, Volker MA, Toomey JA, Lee GK,

Smerback AM, et al.Randomized clinical trial replication of

a psychosocial treatment for children with high-functioning

autism spectrum disorders. Psychology in the Schools 2012;

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Turner-Brown 2008 {published data only}

Turner-Brown LM, Perry TD, Dichter G, Bodfish J,

Penn DL. Brief report: feasibility of social cognition and

interaction training for adults with high-functioning autism.


1777–84.

Wellman 2002 {published data only}

Wellman HM, Baron-Cohen S, Caswell R, Gomez JC,

Swettenham J, Toye E, et al.Thought-bubbles help children

with autism acquire an alternative to a theory of mind.

Autism 2002;6(4):343–63.

Whalen 2003 {published data only}

Whalen C, Schreibman L. Joint attention training

for children with autism using behavior modification

procedures. Journal of Child Psychology and Psychiatry 2003;

44(3):456–68.

White 2010 {published data only}

White SW, Koenig K, Scahill L. Group social skills

instruction for adolescents with high-functioning

autism spectrum disorders. Focus on Autism and OtherDevelopmental Disabilities 2010;25(4):209–19.

Wood 2009 {published data only}

Wood JJ, Drahota A, Sze K, Dyke M, Decker K, Fujii C,

et al.Brief report: effects of cognitive behavioral therapy on

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of two prelinguistic communication interventions on the

acquisition of spoken communication in preschoolers with

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spectrum disorders. Journal of Consulting and Clinical

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References to studies awaiting assessment

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∗ Indicates the major publication for the study



C H A R A C T E R I S T I C S O F S T U D I E S

Characteristics of included studies [ordered by study ID]

Baghdadli 2013

Methods RCT. Control condition: structured group leisure activities

Participants High-functioning autism

Aged eight to12 years

All male

Intellectual level - normal range. Participants matched on verbal IQ

Diagnosis by clinical best estimate, confirmed by ADOS and ADI-R

Final n = 13 (93%)

Interventions Designed to teach ToM and precursor skills

Targets emotion recognition from faces, conversation, social problem solving, stress

management, and ToM

Delivery in group treatment sessions with two therapists. Methods include role play,

video-modelling and problem solving skills. Based on manual of treatment goals includ-

ing techniques, learning stages and tools

Dose: 90 minutes per week, 20 weeks spread over six months

Outcomes Primary outcome: DANVA-2 short form (emotion recognition assessment)

Secondary measures: KidScreen, parent-report measure of quality of life

Notes This study was partially funded by the Pfizer Foundation.

Risk of bias

Bias Authors’ judgement Support for judgement

Random sequence generation (selection

bias)

Low risk Computerised random number generator

Allocation concealment (selection bias) Unclear risk List given to therapists, though allocation

did take place after baseline assessment


(performance bias)

All outcomes

High risk “Until the end of the study, none of the par-

ticipants (children and parents) were told

which group they were assigned to” (p. 436)

. However not possible to blind them to

group activity content

Blinding of outcome assessment (detection

bias)

All outcomes

Low risk Assessors of outcome measures were

blinded



Baghdadli 2013 (Continued)

Incomplete outcome data (attrition bias)

All outcomes

Low risk Use Intention-to-treat analysis as well as as-

sessment of study-completers

Selective reporting (reporting bias) Low risk No evidence of selective reporting

Other bias Low risk Used independent fidelity coding system to

ensure treatment adherence by therapists

Begeer 2011

Methods RCT; wait-list control

Participants All forms of ASD

Aged eight to13 years old

Both genders

Intellectual level: normal range. Participants matched on chronological age, full-scale

IQ, verbal IQ, and non-verbal IQ

Diagnosis by clinical best estimate confirmed by SRS and ASQ

Final n = 36 (90%)

Interventions Designed to teach ToM

Target ToM understanding

Delivered by manualised small group training, plus monthly parental training and

homework. Sessions supervised by certified therapists. Training progresses through 53

structured settings from precursors of ToM (e.g. recognising emotions and intentions),

through elementary skills (e.g. deception, understand others’ mental states) to complex

ToM skills (e.g. second order mental state reasoning)

Dose: one and a half hours weekly for 16 weeks

Outcomes Primary outcome: The ToM test.

Secondary measures: Levels of Emotional Awareness Scale - Children; self-reported em-

pathy; parent-report social behaviours

Notes Funding source not reported.

Risk of bias



bias)

Unclear risk “An independent researcher made the allocation schedule”, p.

1000

Allocation concealment (selection bias) Unclear risk Insufficient information available


(performance bias)

All outcomes

High risk Not possible to blind personnel and participants



Begeer 2011 (Continued)


bias)

All outcomes

Unclear risk No description of outcome assessors


All outcomes

Unclear risk Four families lost to follow-up, three from control group: “re-

fused to return for testing”, p. 1000

Selective reporting (reporting bias) Low risk No evidence of selective reporting

Other bias Low risk

Bolte 2002

Methods RCT: wait-list control

Participants HFA and AS

Aged 16 to 40 years

All male

Intellectual level: normal range. Participants matched on non-verbal IQ

Diagnosis confirmed by ADOS & ADI-R

Final n = 10 (100%)

Interventions Designed to teach precursor skills of ToM

Targets affect recognition from face and eyes

Delivered by “FEFA” computer program: presents six basic emotions plus neutral faces,

users match faces or pictures of eyes to the correct written label

Dose: two hours per week for five weeks

Outcomes Primary outcome measures: Built-in test condition and IAPS (International Affective

Picture System) ratings

Secondary outcome: fMRI

Notes Bolte 2006 - second report of same data set.

This study was funded by the German Research Foundation (Deutsche Forschungsge-

meinschaft, DFG)

Risk of bias



bias)

Low risk Participants pooled and numbered randomly, then allocated

alternately to each group (information provided by email)

Allocation concealment (selection bias) Low risk Participants assigned to group after initial assessment, using

procedure described above



Bolte 2002 (Continued)


(performance bias)

All outcomes

High risk Participants and personnel not blind


bias)

All outcomes

Unclear risk Outcome assessors not blind, but primary outcomes measures

computer administered (p. 65)


All outcomes

Low risk No evidence of attrition

Selective reporting (reporting bias) High risk IAPS data not reported in either publication; author attempted

to locate these scores but no longer available

fMRI loss of data in 2006 report unexplained.

Other bias Low risk

Fisher 2005

Methods RCT; executive function training comparison group, also TAU control but non-random

selection for this group

Participants ASD and AS.

Aged six to 15 years old.

Both genders.

Intellectual level: low range. Participants matched on verbal IQ and non-verbal IQ raw

scores

Diagnosis based on clinical best estimate plus DSM-IV checklist

Final n = 27 (100%).

Interventions Designed to teach ToM.

Targets false belief-skills.

One-to-one training by therapists, taught a strategy of thinking about beliefs as “photos in

the head”, and using illustrative stories. Progress across five stages of increasing complexity

existence of mental states to comprehending false belief

Dose: up to a maximum of 10 x 25mins (p. 763).

Outcomes Primary Outcome: ToM ability, measured by false belief and other tasks, including

teacher-report measure

Notes This study was funded by the Medical Research Council, UK.

Risk of bias




Fisher 2005 (Continued)


bias)

High risk Participants in control group selected by

non-attendance at school during training

period. Allocation to ToM and executive

function intervention groups may be ran-

dom, but no method detail provided (p.

759). Also “children from the same school

were distributed across groups, to control

for any school effects” (p. 759)

Allocation concealment (selection bias) High risk Not addressed in the report, but control

group not randomly assigned


(performance bias)

All outcomes

High risk Impossible to conceal intervention type

from therapist or participant but partici-

pants probably not aware of link between

intervention type and different outcome

measures


bias)

All outcomes

High risk Unclear but seems likely that outcome tests

were administered by therapists


All outcomes

Unclear risk Five teacher questionnaires are missing (p.

759) attributed to the fact that they had too

many to complete each day

Selective reporting (reporting bias) Low risk No evidence of selective reporting.

Other bias Low risk

Golan 2006

Methods RCT; TAU control.

Participants HFA & AS.

Aged 17 to 52 years.

Both genders.

Intellectual level: normal range. Participants matched on chronological age, full-scale,

verbal and non-verbal IQ

Diagnosis confirmed by AQ.

n = 41 (89%).

Interventions Designed to teach a precursor skill of ToM.

Targets complex emotion recognition.

Delivered by MindReading software at home: an emotion library, trainer, and games all

promote understanding of how photographs and film clips of facial emotions match on

to vocabulary and emotional stories

Dose: required minimum use of 10 hours over a 10-week period



Golan 2006 (Continued)

Outcomes Primary outcome: Emotion recognition using a range of computerised tests

Notes This study was funded by multiple contributors comprising: the National Alliance for

Autism Research, the Corob Charitable Trust, the Cambridge Overseas Trust, the B’nai

B’rith Leo Baeck scholarships, the Shirley Foundation, the Medical Research Council

and the Three Guineas Trust

Risk of bias



bias)

Unclear risk States random allocation but not described.

Allocation concealment (selection bias) High risk Not described directly but report implies randomisation oc-

curred before initial assessment


(performance bias)

All outcomes

High risk Participants not blind but unaware of intervention goals of the

study


bias)

All outcomes

Low risk Three assessors were blind to group but one (the lead author) was

not. Outcome measures were computer-presented and therefore

resilient to bias


All outcomes

High risk Five participants in the intervention group dropped out due to

not finding time to complete the required amount of work (p.

597). Study may over-estimate value of intervention

Selective reporting (reporting bias) Low risk One participant’s data not reported for one of many outcome

measures (p. 602, Table 2)

Other bias Low risk

Golan 2010


Participants ASD.

Aged four to eight years.

Both genders.

Intellectual level: normal range. Participants matched for chronological age and verbal

IQ

Diagnosis confirmed by ADI-R and CAST.

Final n = 38 (97%).



Golan 2010 (Continued)

Interventions Designed to teach precursor skills of ToM

Targets emotion comprehension

Delivered by Transporters cartoon on DVD watched at home: cartoon episodes depict

vehicles with real human faces who experience emotional situations and display appro-

priate facial reactions

Dose: recommend three episodes per day for four weeks.

Outcomes Primary Outcomes: Emotion vocabulary and emotion recognition from complex situa-

tions

Notes This study was funded by Culture Online and the UK Department for Culture, Media

and Sport

Risk of bias



bias)

Unclear risk States random allocation but procedure not described.

Allocation concealment (selection bias) Unclear risk Timing and method not described.


(performance bias)

All outcomes

High risk Not possible to blind participants.


bias)

All outcomes

Unclear risk Blinding of outcome assessors not reported, but computer pre-

sentation of outcomes measures resistant to bias


All outcomes

Low risk Only one participant dropped out (control group) but this is

unexplained

Selective reporting (reporting bias) Low risk No evidence of this.

Other bias Low risk

Goods 2013

Methods RCT: TAU control (regular school program, 30 hrs per week).

Participants Autism diagnosis.

Aged three to five years.

No gender information provided.

Intellectual level: low range. Participants matched on chronological age, full-scale IQ,

and verbal IQ

Clinical best estimate diagnosis confirmed by ADOS.

Final n = 11 (73%).



Goods 2013 (Continued)


Targets joint engagement and social communication skills.

Delivered by individual therapists sessions: children taken out of regular classroom ac-

tivities. Approach also described in Kasari 2006 & Kasari 2010.

Dose: Two sessions of 30 minutes each per week, for 12 weeks intervention period

Outcomes Primary outcomes: Early Social Communication Scales, Structured Play Assessment,

classroom observation of joint engagement, and spontaneous communicative gestures

Notes This study was funded by the Organisation for Autism Research and Autism Speaks

Risk of bias



bias)

Low risk Blocked randomisation using SPSS ran-

dom number generator.

Allocation concealment (selection bias) High risk Alllocation post-assessment but conceal-

ment not described. As the blocks were of

fixed length the final allocation within each

block would be known


(performance bias)

All outcomes

High risk Not possible to blind participants or per-

sonnel.


bias)

All outcomes

Low risk Assessors blinded for all outcomes.


All outcomes

High risk Attrition four out of 15 participants. Anal-

ysis considered completers only

Selective reporting (reporting bias) High risk One measure (Reynell Developmental

Language Scales) reported in Table 2 (no

group differences at Baseline or Exit) but

not discussed

Other bias Low risk



Hadwin 1996

Methods RCT: three intervention groups (emotion, belief, play).

Participants Autism.

Aged four to 13 years.

Both genders.

Intellectual level: mostly low range, but four children with verbal mental age in normal

range (distributed across groups). Participants matched on chronological age, verbal

mental age, and expressive language

Diagnosis confirmed by DSM-III and DSM-IV checklist (see Hadwin 1997, p. 524).

Final n = 30 (100%).


Three intervention groups each targeting a specific ToM component: Understanding

Emotion, Understanding Belief, and Pretend Play

Delivered by one-to-one therapist led intervention: used a question and answer structure

with corrective feedback. Play session additional involved modelling of pretend play.

Good performance on each level required for progress to the next level of complexity

Dose: One assessment session, then eight therapy sessions, followed by one post-test

assessment session. Eight, consecutive daily half-hour therapeutic sessions

Outcomes Primary outcome: teaching level reached on five-point scale.

Secondary outcomes: generalisation of taught skills to novel materials. Conversation and

use of mental state language in a story-book task (Hadwin 1997).

Notes also see Hadwin 1997.

This study was funded by the Bethlem-Maudsley Research Fund and the Mental Health

Foundation

Risk of bias



bias)

Unclear risk States random allocation but procedure not

described.

Allocation concealment (selection bias) High risk Baseline assessment conducted after alloca-

tion to treatment condition


(performance bias)

All outcomes

High risk Not possible to blind participants or per-

sonnel.


bias)

All outcomes

High risk Not explicitly stated, but seems that both

pre- and post-test data were collected by

unblinded therapists


All outcomes

Low risk No evidence of attrition.



Hadwin 1996 (Continued)


Other bias Low risk

Hopkins 2011

Methods RCT: control group with art and design computer program.

Participants ASD.

Aged six; three to 15; one years.

Both genders.

Intellectual level: 24 in the normal range (HFA), 25 in a low-functioning range (LFA).

Participants matched on chronological age, verbal IQ and non-verbal IQ

Diagnosis by clinical best estimate confirmed with CARS.

Final n = 49 (96%).

Interventions Teaches precursor skills of ToM.

Targets emotion recognition, eye contact and facial identity recognition

Delivered by FaceSay: social skills training via a computer program: three games to:

match gaze direction with an object; select parts of faces which fit with a whole face; and

match facial expressions with emotions

Dose: 12 x 30 minute sessions over 6 weeks.

Outcomes Primary outcome: Emotion recognition from photos and drawings of faces and Benton

Face Recognition Test

Secondary outcomes: Social Skills Rating System, Social skills observation

Notes Authors were emailed for further information, no response.This study was partially funded by a grant from Civitan International and with the co-

operation of Symbionica LLC

Risk of bias



bias)

Unclear risk No information provided.

Allocation concealment (selection bias) Unclear risk No information provided.


(performance bias)

All outcomes

Unclear risk Parents and participants technically blind

but could have guessed from computer pro-

gram content


bias)

All outcomes

Low risk SSRS: observed by blinded raters.

Other measures resistant to bias.



Hopkins 2011 (Continued)


All outcomes

Low risk Two children excluded due to low atten-

dance, and reasons given


Other bias Low risk

Ingersoll 2012


Participants Core autism.

Aged 27 to 47 months old.

Both genders.

Intellectual level: low range. Participants matched on chronological age, non-verbal men-

tal age, and language age

Diagnosis based on ADOS and clinical best estimate.

Final n = 27 (93%).

Interventions Designed to teach precursor skills of ToM.

Targets imitation skills.

Therapist-delivered reciprocal imitation training: naturalistic play-based imitation in-

tervention using pairs of identical play materials to model social imitation skills. Actions

are modelled and paired with a verbal marker with physical prompts used where children

do not respond

Dose: three hours per week for 10 weeks plus.

Outcomes Primary outcomes: Early Social Communication Scales, initiation of joint attention.

Social Emotional Scale of the Bayley Scales of Infant Development 3rd Edition, parent-

report

Secondary (mediator) measures: Elicited and spontaneous imitation skills (motor imita-

tion, unstructured imitation assessment)

Notes See also Ingersoll 2010.

Funding source not known.

Risk of bias



bias)

Low risk Randomised by a coin toss.

Allocation concealment (selection bias) Low risk Pairing on expressive language age before assignment by coin

toss



Ingersoll 2012 (Continued)


(performance bias)

All outcomes

High risk Not possible to blind participants and therapists.


bias)

All outcomes

Low risk In Ingersoll 2010, p. 1155 “Assessments were scored by trained

research assistants blind to group” Confirmed in personal com-

munication: although examiners were not blind, coders were


All outcomes

Low risk Two children withdrew, and reasons given. Analysis of com-

pleters only


Other bias Unclear risk No reason given for adding additional subjects to Ingersoll 2010,

to randomisation may not be secure

Kaale 2012

Methods RCT: TAU control.

Participants Autism and ASD.

Aged 29 to 60 months.

Both genders.

Intellectual level: low range. Participants matched on chronological age, mental age,

expressive and receptive language ages, and developmental level

Diagnosis confirmed by ADOS (or ADI for 49 participants).

Final n = 61 (100%).


Targets joint attention and joint engagement.

Delivered as manualised Joint attention intervention, by pre-school teachers: table top

training sessions provide multiple opportunities for a child to initiate a targeted skill.

Skills are encouraged using verbal and physical prompts, presenting opportunities and

positive feedback

Dose: two daily sessions, five days per week for eight weeks

Outcomes Primary outcome: ESCS.

Secondary outcome: Teacher-child play and mother-child, both rated for joint attention

and joint engagement

Notes This study was funded by the South-Eastern Norway Regional Health Authority and

the Centre for Child and Adolescent Mental Health (Eastern and Southern Norway)

Risk of bias




Kaale 2012 (Continued)


bias)

Low risk Concealed randomisation list generated before the study began

Allocation concealment (selection bias) Low risk Allocation after baseline assessment.


(performance bias)

All outcomes

High risk Parents and pre-school staff were blind to allocation at baseline

(p. 98), but therapists were not blind (p. 101). Participants likely

to be unaware of intervention


bias)

All outcomes

Low risk Blinded coders and outcome assessors (p. 98).


All outcomes


Selective reporting (reporting bias) Low risk All pre-specified outcomes were reported.

Other bias Low risk

Kasari 2006

Methods RCT; two control groups: TAU and symbolic play.

Participants ASD and core autism.


Both genders.

Intellectual level: low range. Participants matched on chronological age, mental age,

developmental level and expressive and receptive language ages

Diagnosis confirmed by ADOS and ADI.

Final n = 58 (89%).


Targets joint attention.

Delivered as one to one therapist led intervention: table top training and then floor

sessions provide multiple opportunities for a child to initiate a targeted skill. Skills are

encouraged using verbal and physical prompts, presenting opportunities and positive

feedback. Structure is withdrawn as the training progresses

Dose: 30 minutes daily for five to six weeks.

Outcomes Primary outcomes: ESCS.

Secondary outcome: parent-child play rating, coded for joint engagement and joint

attention

Notes See also:

Gulsrud 2007: sub-analysis of n = 35 with age range 33 to 54 months.

Arora 2008: PhD thesis reporting on links between joint attention and perseveration for

n = 35



Kasari 2006 (Continued)

Kasari 2008: specific report on language outcome for the full sample, Reynell scales of

language development

Kasari 2012b: longitudinal follow-up, reporting on expressive vocabulary and Differen-

tial Abilities Scale

This study was funded by the National Institute for Health and the CPEA network

Risk of bias



bias)

Low risk “used generated numbers from computer

program” (information provided by au-

thor)

Allocation concealment (selection bias) High risk “Families who consented were randomised

and began treatment right after assessments

(by outside independent assessors” and “al-

location to group was done as kids entered

the Early Intervention Program and parent

consented - next allocation of group num-

ber was revealed after eligibility and con-

senting” (information provided by author)

. However the published report states that

some children were excluded because they

did not meet inclusion criteria (p. 612) in-

dicating that assessments were completed

but not scored before allocation


(performance bias)

All outcomes

High risk Not possible to blind parents, children or

therapists, though children likely to have

been unaware


bias)

All outcomes

Low risk Outcome assessors and coders both blind

to group (information provided by author)


All outcomes

Unclear risk Seven participants did not meet criteria, re-

fused final assessment or left the program

unexpectedly. Distributed across groups

(Joint attention intervention = two; sym-

bolic play intervention = one; control group

= four)

Selective reporting (reporting bias) Unclear risk No evidence of selective reporting.

Other bias Low risk



Kasari 2010

Methods RCT: wait-list control.



Both genders.

Intellectual level: low range. Participants matched on chronological age, mental age, and

developmental level

Diagnosis by clinical best estimate and ADI.

Final n = 35 (83%).


Targets joint attention and joint engagement.

Delivered as parent-mediated training adapted from Kasari 2006. Core principles form

10 modules, delivered by parents trained by therapists. Uses aspects of applied behaviour

analysis and facilitative and responsive interaction to encourage target behaviours in the

child

Dose: three modules per week for eight weeks.

Outcomes Primary outcome: Videotaped parent-child interaction, coded for engagement, type of

play, and frequency of joint attention

Secondary outcomes: caregiver quality of involvement, adherence to treatment, and

service utilisation measure

Notes This study was funded by the National Institute for Mental Health

Risk of bias



bias)

Low risk Random numbers list.

Allocation concealment (selection bias) Low risk Children randomised after meeting study criteria.


(performance bias)

All outcomes

High risk Not possible to blind parents and participating children.


bias)

All outcomes

Low risk Examiners and coders both blind to group status.


All outcomes

Low risk Two children did not meet criteria, two parents declined to

participate. Three further children from the control group de-

scribed as “did not receive allocated intervention”. Though not

explicitly stated it appeared from Table 4 that analyses were

based on intention-to-treat rather than treatment completers

alone

Selective reporting (reporting bias) Low risk No evidence of selective reporting (pp. 1052-1053).



Kasari 2010 (Continued)

Other bias Low risk

Kim 2009

Methods RCT with cross-over; toy play session control.



Final sample all male.

Intellectual level: normal range. Participants not matched but evaluated for developmen-

tal level

Diagnosis confirmed by Korean CARS and four received ADOS as well

Final n = 10 (66%).


Targets joint attention.

Delivered as improvised music sessions based on a semi-flexible treatment manual. Ses-

sions included free play time and directed activities with therapist modelling turn-taking

and other activities

Dose: 12 weeks, 30 minutes per week.

Outcomes Primary outcomes: PDD-BI social approach subscale, and ESCS.

Secondary outcomes: observational coding of emotional and motivation responsiveness,

and responsiveness to joint attention / joint attention initiation

Notes See also Kim 2008 for details of PDD-BI and ESCS outcome measure.

Email sent to request more information but none received.This study was funded by Aalborg University, Denmark.

Risk of bias



bias)

Unclear risk “participants were randomly assigned to

two groups” - but no details given

Allocation concealment (selection bias) Unclear risk No details given.


(performance bias)

All outcomes

High risk Not possible to blind participants and per-

sonnel.


bias)

All outcomes

Low risk Coders for PDD-BI and ESCS both blind.

PDD-BI administered to both blind teach-

ers and unblinded parents, so in the latter

case, susceptible to bias even if the inter-

viewer is blind

30% of observational behaviour measures



Kim 2009 (Continued)

second-coded by blind coder for reliability


All outcomes

Unclear risk Five children dropped out due to ill health

or long distances to travel to receive inter-

vention


Other bias Low risk Differences in ratings provided by moth-

ers and professionals. Professional ratings

support efficacy of intervention and these

were also blind. Mothers over-estimate pre-

intervention abilities and thus under-esti-

mate efficacy of the intervention, but they

were not blind (in Kim, 2008, p. 1763)

Landa 2011

Methods RCT: contact (non-interpersonal synchrony) control.



Both genders.

Intellectual level: low range. Participants matched on chronological age, receptive lan-

guage and visual reception standardised scores

Diagnosis confirmed by ADOS.

Final n = 48 (96%).


Targets socially synchronous behaviour.

Delivered by trained teacher using the Assessment Evaluation Program System (AEPS)

for infants and children. Instructional strategies ranged from discrete trial teaching to

pivotal response training and routine-based interactions. Intervention incorporated low-

tech communication systems. Highly motivating tasks and materials designed to elicit

frequent child-initiated communication

Dose: two and a half hours per day, four days per week for six months in classroom

Additional one and a half hours per month parent training and 38 hours parent education

Outcomes Primary Outcome: Socially engaged imitation in a structured imitation task

Secondary outcomes: Initiations of joint attention & shared positive affect - measured

using the CSBS; Mullen Scales of Early Learning expressive language and visual reception

subscales

Notes This study was funded by the National Institute of Mental Health and HRSA

Risk of bias




Landa 2011 (Continued)


bias)

Unclear risk No information provided.

Allocation concealment (selection bias) Unclear risk Participants placed into matched pairs

prior to randomisation which could intro-

duce bias


(performance bias)

All outcomes

Low risk Parent, children and teachers all blind to

group: not possible to blind intervention

trainers


bias)

All outcomes

Low risk Assessment at baseline and follow-up by a

independent, blind clinician (p. 16)


All outcomes

Low risk One family withdrew from the study, and

one was not analysed due to missing base-

line data


Other bias Low risk

Ryan 2010

Methods RCT: wait-list control.


Aged six to 14 years.

Both genders.

Intellectual level: normal range. Participants matched on verbal IQ and non-verbal IQ

Diagnosis confirmed by ADOS and DISCO.

Final n = 30 (100%).


Targets emotion recognition.

Delivered as therapist-led training using photos of six core facial emotions. Thera-

pists highlighted component parts of emotional expressions (e.g. raised eye-brows) and

matched faces with verbal labels. Participants engaged in matching games, role-play,

tracing, and drawing of faces to support direct instruction

Dose: one hour per week over four weeks.

Outcomes Primary outcome: Facial emotion recognition test.

Notes This study was funded by the COPE Foundation and supported by the Catherine T

MacArthur Foundation Research Network on Early Experience and Brain Development

Risk of bias



Ryan 2010 (Continued)



bias)

Low risk “We assigned sequential numbers to each child (ordered al-

phabetically) to be included in the study and then used the

random number table provided in David Clark-Carter’s book

”Doing Quantitative Psychological Research - From Design to

Report“ (1997) to form two groups” (information provided

by author)

Allocation concealment (selection bias) High risk Assessments conducted before allocation, but using open ran-

dom numbers table could have introduced bias


(performance bias)

All outcomes

High risk Not possible to blind personnel and participants.


bias)

All outcomes

Unclear risk Independent outcome assessor was “ blind to the initial scores

achieved by each of the children, to the membership of the

treatment groups and to our hypotheses” (information pro-

vided by author)


All outcomes

Low risk No evidence of attrition at four weeks, immediately post in-

tervention. However five children lost to follow-up at three

months, after all had received the training, without reason

given

Selective reporting (reporting bias) Low risk No evidence of selective reporting, but only one outcome mea-

sure was used

Other bias Low risk

Schertz 2013

Methods RCT: TAU control.

Participants Autism spectrum disorder and autism.

Aged under 30 months (mean age in each group: 24.6 months and 27.5 months)

Gender information not reported.

Intellectual level: normal range. Participants matched on chronological age, receptive

and expressive language

Diagnosis confirmed by ADOS and M-CHAT (to establish high risk)

Final n = 23 (100%, but see risk of bias below).

Interventions Designed to teach precursor skill of ToM.

Targets joint attention and sub-skills (Focus on Faces, Turn-Taking)

Delivered as parent-training delivered by intervention co-ordinators at home. Sessions

included guidance and reflection based on filmed segments of parent-child play, and a

manual for parents



Schertz 2013 (Continued)

Dose: Parent training sessions were weekly at home for at least 15 sessions. Parents agree to

spend 30 minutes per day in face-to-face interaction with their child. Total intervention

period ranged from 4 to 12 months (mean 7 months)

Outcomes Primary outcomes: Precursors of Joint Attention Measure (PJAM), based on coding of

parent-child interaction

Secondary measures: Vineland Adaptive Behaviour Scales and Mullen Scales of Early

Learning

Notes This study was funded by Autism Speaks.

Risk of bias



bias)

Unclear risk Once entry criteria were met, children were paired and then

assigned to intervention or control. Randomisation was by coin

toss (confirmed by author email)

Allocation concealment (selection bias) High risk No information given, but coin toss open to abuse if performed

by intervention co-ordinators


(performance bias)

All outcomes

High risk Not possible to blind parents and intervention co-ordinators


bias)

All outcomes

Low risk Intervention co-ordinators and parents carried out assessments

but these were coded by raters blind to group (but not time

point)


All outcomes

Low risk There is unreported attrition of one participant (confirmed by

author)

Selective reporting (reporting bias) Unclear risk No evidence of selective reporting.

Other bias High risk The treatment period was of variable length. Treatment was ter-

minated (and outcomes measures taken) once three or more

instances of initiation of joint attention were observed across

multiple sessions. Within each pair, inter-assessment period was

identical, but this variable system means that 100% of partici-

pants made gains in responses to treatment. i.e. the treatment

effect was weighted by this study design feature



Solomon 2004

Methods RCT, wait-list control.

Participants HFA, AS and PDD-NOS.

Aged eight to 12 years.

All male.

Intellectual level: normal range, Participants matched on chronological age and full-scale

IQ

Diagnosis confirmed by ADOS and ADI.

Final n = 18 (100%).


Targets social adjustment, emotion recognition, ToM, and executive functions

Delivered as group social skills training sessions with parent psychoeducational sessions.

Children’s group sessions followed a strict structure, including welcome, lesson time,

games and free social time. The curriculum followed 10 modules focusing on emotional

understand and empathy, conversational skills, and lessons about friendship

Dose: one and a half hours per week for 20 weeks.

Outcomes Primary outcomes: Diagnostic Analysis of Non-Verbal Accuracy - facial expression recog-

nition subscale; Happe’s Strange Stories; The Faux-Pas Recognition Test; Test of Problem

Solving (TOPS)

Secondary outcomes: Depression Inventories for children and parents; problem be-

haviour logs

Notes Authors were emailed for further information but no response received.This study was funded by the MIND Institute, UC Davis, California, USA

Risk of bias



bias)

Low risk Participants were matched into pairs and then “Nine boys were

randomly selected... to serve as the first intervention group

subjects” - but method unclear (p. 654)

Allocation concealment (selection bias) Unclear risk Participants assessed and matched before random allocation,

but pairing process could have introduced bias


(performance bias)

All outcomes

High risk Not possible to blind participants and personnel.


bias)

All outcomes

High risk Unlikely to have been blinded as assessments carried out by

first author


All outcomes




Solomon 2004 (Continued)


Other bias Low risk

Williams 2012

Methods RCT; control group Thomas the Tank Engine DVD.

Participants Autism spectrum disorder, including autism.

Aged four to seven years.

Intellectual level: low to normal range, but cognitive level above 30 months minimum.

Participants matched on chronological age, full-scale IQ, verbal IQ and non-verbal IQ

Diagnosis based on clinical best estimate confirmed by ADOS.

n = 55 (92%).



Delivered by Transporters DVD: cartoon episodes depict vehicles with real human faces

who experience emotional situations and display appropriate facial reactions

Dose: 15 minutes per day for four weeks (mean 11.76 hours total)

Outcomes Primary outcomes: NEPSY-II affect recognition test using pictures, and the Pictures of

Facial Affect test (using Ekman faces: identify emotions from photos of faces)

Secondary measures: NEPSY-II ToM tasks; Mindreading tasks; Vineland Adaptive Be-

haviour Scales (socialisation subscale)

Notes This study was funded by the Financial Markets Foundation for Children, Australia

Risk of bias



bias)

Low risk Satistician randomised the allocation.

Allocation concealment (selection bias) Low risk Each DVD packed in an unmarked num-

bered envelope - adequate concealment


(performance bias)

All outcomes

High risk Participants not blind to DVD content.


bias)

All outcomes

Low risk Assessor blind to treatment group.



Williams 2012 (Continued)


All outcomes

High risk Attrition of five participants after one

month (unbalanced between groups) and

only completers analysed. Missing data on

emotion identification where children (n =

eight) could not complete the task: these

may relate to outcome. Effect sizes not

given. Attirition of nine more participants

at three-month follow-up (unbalanced be-

tween groups) so follow-up data used last

observation carried forward


Other bias Low risk

Wong 2010

Methods RCT cross-over: wait-list control.

Participants ASD.


Both genders.

Intellectual level: low range. Participants matched on chronological age and mental age

Diagnosis confirmed by ADI-R, ADOS and CARS.

Final n = 17 (100%).


Targets eye contact, gesture and vocalisation.

Delivered as direct one to one therapy: modelling gestures accompanied by verbal labels,

eliciting requests using favourite toys

Dose: 10 x 30 minute sessions over two weeks.

Outcomes Primary outcome: ADOS (reported item by item).

Secondary outcomes: Ritvo-Freeman Real Life Rating Scale (RFRLRS) - parent rated;

Symbolic Play Test; Parenting Stress Index, short form

Notes Authors emailed for more detail but no reply received.Funding source unknown.

Risk of bias



bias)

High risk “17 children consecutively diagnosed were random-

ized into the Intervention and Control groups” - as-

signment may have been alternate rather than ran-

dom? (p. 679)



Wong 2010 (Continued)

Allocation concealment (selection bias) High risk Baseline assessment carried out after randomisation

to group


(performance bias)

All outcomes

High risk Not possible to blind participants or personnel.


bias)

All outcomes

Low risk ADOS and SPT both administered and scored by

blind assessors. Some outcomes are parent-reported

measures and therefore unblinded. (pp. 680-681)


All outcomes



Other bias Unclear risk Wait-list control group combined with Interven-

tion group to give larger sample size for analysis

Wong 2013

Methods Cluster-RCT: repeated measures, nested within children, nested within classrooms. Con-

trol conditions: symbolic play intervention, wait-list control

Participants Autism.

Aged three to six years.

Both genders.

Intellectual level: low range. Participants matched on chronological age, mental age, and

expressive and receptive language ages

Diagnosis based on clinical best estimate and confirmed by CARS

Final n = 33 (97%).


Targets joint attention and interpersonal engagement behaviours

Delivered as teacher training. Teacher then implemented the approach in the classroom

according to their own preference (e.g. one-to-one, small group, whole class)

Dose: teacher training sessions were one hour per week, for four weeks

Outcomes Primary outcome: Early Social Communication Scales, and Structured PLay Assessment.

Also direct classroom observation of child and teacher behaviours (joint engagement,

joint attention)

Secondary measures: teacher acceptability of intervention.

Notes Funding source unknown.

Risk of bias



Wong 2013 (Continued)



bias)

Low risk Teacher assignment to one of three groups

by random numbers list

Allocation concealment (selection bias) Low risk Allocation occurred before initial assess-

ment, but allocation list concealed from

baseline assessors (confirm by author email)


(performance bias)

All outcomes

High risk Teachers not blinded, as recipients of train-

ing.


bias)

All outcomes

Low risk Research observers for all measures were

blinded (confirmed by author email)


All outcomes

Low risk One teacher (and therefore one child)

dropped out; reason given


Other bias Low risk

Young 2012

Methods RCT; control group Thomas the Tank Engine DVD.

Participants All forms of ASD.

Aged four to eight years.

Intellectual level: normal range. Participants matched on verbal and non-verbal IQ

Diagnosis based on clinical best estimate confirmed by SCQ.

Final n = 25 (100%).



Delivered by Transporters DVD: cartoon episodes depict vehicles with real human faces

who experience emotional situations and display appropriate facial reactions

Dose: five to 10 minute episodes, children watch three per day for three weeks

Outcomes Primary outcome: Social Communication Questionnaire, parent-report

Secondary outcomes: NEPSY-II affect recognition subscale using pictures; The Faces

Task - emotion recognition from photos of faces

Notes Funding source unknown.

Risk of bias



Young 2012 (Continued)



bias)

High risk “Random allocation was done by sim-

ply alternating between the 2 interven-

tion groups for each participant as they re-

sponded to the letters which were sent out .

.. we tossed a coin to see which group went

first and it was Thomas and then the next

person who responded was Transporters

and so on” (information from the author)

Allocation concealment (selection bias) High risk Published report states that assignment was

after baseline assessment (p. 987) but au-

thor’s personal communication states that

“random allocation occurred before base-

line assessment- when the participant reg-

istered interest in the study they were allo-

cated a condition”


(performance bias)

All outcomes

Low risk Parents and children aware of DVD con-

tent but “ they were naive to the purpose of

the study” (information from the author)


bias)

All outcomes

Unclear risk “assessor was not blind to treatment but the

measures didn’t require any subjective in-

terpretation. Parents completed question-

naires and they were naive to the purpose of

the study” (information from the author)


All outcomes


Selective reporting (reporting bias) Low risk All pre-specified outcomes are reported. So

too are four outcome measures not pre-

specified, nor described in the methods

Other bias Low risk

ADI-R: Autism Diagnostic Interview - Revised

ADOS: Autism Diagnostic Observation Schedule

AS: Asperger’s syndrome

ASD: autism spectrum disorder

ASQ: autism spectrum quotient

CARS: Childhood Autism Rating Scale

DANVA-2: Diagnostic Analysis of Non-Verbal Accuracy

DISCO: Diagnostic Interview for Social and Communication Disorders

DSM: Diagnostic and Statistical Manual of Mental Disorders

ESCS: Early Social Communication Scales



fMRI: functional magnetic resonance imaging

HFA: high-functioning autism

ICD-10: International Classification of Diseases (10th revision)

LFA: low-functioning autism

M-CHAT: Modified Checklist for Autism in Toddlers

NEPSY-II: developmental NEuroPSYchological assessment, 2nd Edition

PDD-BI: Pervasive Developmental Disorder Behavior Inventory

PDD-NOS: Pervasive Developmental Disorder - Not Otherwise Specified

RCT: randomised controlled trial

SCQ: Social Communication Questionnaire

SPT: Symbolic Play Test

SRS: Social Responsiveness Scale

ToM: Theory of Mind

Characteristics of excluded studies [ordered by study ID]

Study Reason for exclusion

Adams 2012 Not ToM-linked

Aldred 2004 Broad-base intervention, not ToM-specific

Beaumont 2008 Broad-base intervention, not ToM-specific

Cardon 2011 Used a multiple baseline case-control design

Carter 2011 Not ToM-linked

Casenhiser 2011 Broad-base intervention, not ToM-specific

Castorina 2011 Not ToM-linked

Charman 2007 No data reported

Corbett 2011 Not ToM-linked

Dawson 2010 Not ToM-linked

DeRosier 2011 Not ToM-linked

Drew 2002 Broad-base intervention, not ToM-specific

Estes 2011 No data reported

Field 2001 Experimental pilot study: not a full intervention trial

Frankel 2010a Not ToM-linked



(Continued)

Frankel 2010b No data reported

Gantman 2012 Not ToM-linked

Gattino 2011 Broad-base intervention, not ToM-specific

Geretsegger 2012 No data reported

Girolametto 1988 Not an ASD sample

Green 2010 Broad-base intervention, not ToM-specific

Hanley-Hochdorfer 2010 Not an RCT/qRCT

Heimann 2006 Experimental pilot study: not a full intervention trial

Hillier 2012 Not an RCT/qRCT

Howlin 1997 No data reported

Howlin 2007 Not ToM-linked

Jocelyn 1998 Not ToM-linked

Jones 2010 Not an RCT/qRCT

Kasari 2012a Not ToM-linked

Kern 2011 Not an RCT/qRCT

Koenig 2010 Not ToM-linked

Lang 2010 No data reported

Laugeson 2012 Not an RCT/qRCT

Lawton 2012 Broad-base intervention, not ToM-specific, teaches play skills as well as joint attention

LeGoff 2004 Not an RCT/qRCT

Lerner 2012 Broad-base intervention, not ToM-specific, teaches a wide range of complex social skills

Lopata 2010 Broad-base intervention, not ToM-specific, teaches a wide range of complex social skills

Mahoney 2005 Not an RCT/qRCT

McConachie 2004 No data reported



(Continued)

McGregor 1998 Not an RCT/qRCT

Minshew 2010 No data reported

Morgan 2010 No data reported

Nefdt 2010 Not ToM-linked

Oosterling 2010 Not an RCT/qRCT

Ozonoff 1995 Not an RCT/qRCT

Pajareya 2011 Broad-base intervention, not ToM-specific

Quirmbach 2009 Not ToM-linked

Roberts 2011 Not ToM-linked

Rodgers 2012 Not ToM-linked

Rogers 2010 Not ToM-linked

Schertz 2007 Not an RCT/qRCT

Schmidt 2012 Not an RCT/qRCT

Silver 2001 ASD diagnosis unconfirmed

Smith 2000 Not ToM-linked

Smith 2004 Not an RCT/qRCT

Stichter 2001 Not an RCT/qRCT

Strain 2011 Not ToM-linked

Swettenham 1996 Not an RCT/qRCT

Tanaka 2010 Not ToM-linked

Temple 2007 No data reported

Thomeer 2012 Broad-base intervention, not ToM-specific, teaches a wide range of complex social skills

Turner-Brown 2008 Not an RCT/qRCT

Wellman 2002 Not an RCT/qRCT



(Continued)

Whalen 2003 Not an RCT/qRCT

White 2010 Not ToM-linked

Wood 2009 Not ToM-linked

Yoder 2006 Broad-base intervention, not ToM-specific

Yoder 2006b Not ToM-linked

ASD: autism spectrum disorder


qRCT: quasi-randomised controlled trial

ToM: Theory of Mind

Characteristics of studies awaiting assessment [ordered by study ID]

Baghdadli 2010

Methods RCT

Participants Fourteen boys aged between eight to 12 years old

Interventions Comparison of the effect of two prosocial interventions (social skill entertainment versus general educative interven-

tion)

Outcomes

Notes Awaiting translation




D A T A A N D A N A L Y S E S

Comparison 1. Treatment effects in meta-analysis

Outcome or subgroup titleNo. of

studies

No. of

participants Statistical method Effect size

1 Joint engagement in

mother-child interaction

2 88 Std. Mean Difference (IV, Random, 95% CI) 0.55 [0.11, 0.99]

2 Emotion recognition from face

photographs, TAU control

4 105 Std. Mean Difference (IV, Random, 95% CI) 0.75 [0.22, 1.29]

3 Joint attention initiations in

standardised assessment

3 92 Std. Mean Difference (IV, Random, 95% CI) 0.23 [-0.48, 0.94]

Analysis 1.1. Comparison 1 Treatment effects in meta-analysis, Outcome 1 Joint engagement in mother-

child interaction.

Review: Interventions based on the Theory of Mind cognitive model for autism spectrum disorder (ASD)

Comparison: 1 Treatment effects in meta-analysis

Outcome: 1 Joint engagement in mother-child interaction

Study or subgroup Experimental Control

Std.Mean

Difference Weight

Std.Mean

Difference

N Mean(SD) N Mean(SD) IV,Random,95% CI IV,Random,95% CI

Kaale 2012 26 57.3 (22.8) 27 49.2 (19.9) 61.6 % 0.37 [ -0.17, 0.92 ]

Kasari 2010 19 42.85 (19.96) 16 27.87 (14.01) 38.4 % 0.84 [ 0.14, 1.53 ]

Total (95% CI) 45 43 100.0 % 0.55 [ 0.11, 0.99 ]

Heterogeneity: Tau2 = 0.01; Chi2 = 1.05, df = 1 (P = 0.30); I2 =5%

Test for overall effect: Z = 2.45 (P = 0.014)

Test for subgroup differences: Not applicable

-10 -5 0 5 10

Favours [control] Favours [intervention]



Analysis 1.2. Comparison 1 Treatment effects in meta-analysis, Outcome 2 Emotion recognition from face

photographs, TAU control.



Outcome: 2 Emotion recognition from face photographs, TAU control

Study or subgroup Experimental Control

Std.Mean

Difference Weight

Std.Mean

Difference


Golan 2006 19 37.5 (7.8) 22 34.8 (8.2) 36.1 % 0.33 [ -0.29, 0.95 ]

Hopkins 2011 13 5 (1) 11 3.31 (1.18) 22.5 % 1.50 [ 0.58, 2.43 ]

Ryan 2010 20 19 (2.49) 12 16.83 (2.37) 29.4 % 0.86 [ 0.11, 1.62 ]

Solomon 2004 4 12.8 (2.9) 4 11.8 (2.4) 12.0 % 0.33 [ -1.08, 1.73 ]

Total (95% CI) 56 49 100.0 % 0.75 [ 0.22, 1.29 ]




-4 -2 0 2 4

Favours control Favours intervention



Analysis 1.3. Comparison 1 Treatment effects in meta-analysis, Outcome 3 Joint attention initiations in

standardised assessment.



Outcome: 3 Joint attention initiations in standardised assessment

Study or subgroup Treatment Control

Std.Mean

Difference Weight

Std.Mean

Difference


Goods 2013 5 0.4 (0.89) 6 1 (1.73) 21.9 % -0.39 [ -1.59, 0.82 ]

Ingersoll 2012 14 4.79 (5.41) 13 0.92 (1.44) 33.7 % 0.93 [ 0.13, 1.73 ]

Kaale 2012 27 1.6 (2.6) 27 1.6 (2.2) 44.3 % 0.0 [ -0.53, 0.53 ]

Total (95% CI) 46 46 100.0 % 0.23 [ -0.48, 0.94 ]




-100 -50 0 50 100

Favours Control Favours Treatment

A D D I T I O N A L T A B L E S

Table 1. Additional methods

Review section Item Methods

Unit of Analysis Cluster-randomised trials Authors will use a summary measure from each

cluster and conduct the analysis at the level of al-

location (that is sample size = number of clusters).

However, if there are very few clusters this would

significantly reduce the power of the trial, in which

case the authors will attempt to extract a direct es-

timate of the risk ratio using an analysis that ac-

counts for the cluster design, such as a multilevel

model, a variance components analysis or gener-

alised estimating equations (GEEs). Statistical ad-

vice will be sought to determine which method is

appropriate for the particular trials to be included

Subgroup Analysis Identification of dimensions for subgroup analysis In future updates the following clinically-relevant

differences may be the focus of subgroup analyses:

1. intervention delivery type (e.g. therapist,

parent-mediated, school-based) and length



Table 1. Additional methods (Continued)

2. intervention target skill (e.g. ToM as a

whole, joint attention, emotion recognition,

false-belief understanding)

3. participant age (e.g. pre-school, young

children, adolescents, adults), IQ (low versus

normal or high), specific diagnosis, and verbal

ability

Sensitivity Analysis Identification of variables for sensitivity analysis In future updates the impact of factors such as high

rates of loss to follow-up or inadequate blinding

on outcomes will be explored

Dealing with Missing Data Procedures for imputation in the event of issues

with missing data

Should unacceptable levels and/or non-random

missing data be found in future studies for in-

clusion in the review, the authors will attempt to

impute missing values. Imputation may use indi-

vidual data (where available from the original re-

port authors) OR group-level summary statistics

(which are normally included in published reports)

. Mean imputation will be used where variables are

normally distributed, and the median will be used

for non-normal distributions. In either case the re-

view will report how the imputed values appear

to change the outcome of the study/meta-analysis

and use this variability to inform the strength of

our conclusions

Table 2. Outcome measures used

Outcome Category Measure Study

Primary Communication (standardised mea-

sure)

Semi-structured conversation task:

telling a story from a picture book

Hadwin 1996

Social Communication Questionnaire

(SCQ)

Young 2012

Autism Diag-

nostic Observation Schedule (ADOS)

: Language and Communication

Wong 2010

Social Function (standardised mea-

sure)

Joint Attention: Early Social Commu-

nication Scale (ESCS)

Kaale 2012

Kasari 2006

Ingersoll 2012

Goods 2013

Kim 2009

Wong 2013



Table 2. Outcome measures used (Continued)

Communication and Symbolic Be-

haviour Scales developmental profile

Landa 2011

Autism Diag-

nostic Observation Schedule (ADOS)

: Reciprocal Social Interaction

Wong 2010

Social Communication Questionnaire

(SCQ)

Young 2012

Social Emotional Scale (SES), Bay-

ley Scales of Infant Development, 3rd

Edition

Ingersoll 2012

Vineland Adaptive Behaviour Scales

(VABS), Socialisation subscale

Williams 2012

Flexibility & imagination (standard-

ised measure)

none

Secondary Intervention specific: ToM ToM test: standardised interview for

Tom understanding

Levels of emotional awareness scale for

children (LEAS-C): performance as-

sessment

Begeer 2011

False-belief tasks (unexpected transfer

and deceptive box): behavioural ToM

task

Penny Hiding Deception Task: be-

havioural ToM task

Seeing Leads to Knowing Task: be-

havioural ToM task

Knowing/Guessing Task: behavioural

ToM task

Fisher 2005

Level of training reached (ToM skills,

pretend play skills, emotion under-

standing)

Generalisations to non-taught tasks

(ToM skills, pretend play skills, emo-

tion understanding)

Generalisation across skill sets and in-

tervention groups (e.g. effects of ToM

intervention on pretend play skills and

so on)

Hadwin 1996

ToM: Strange Stories and Faux Pas

Recognition Test

Solomon 2004




NEPSY-II ToM task Williams 2012

Intervention specific: emotion recog-

nition

FEFA test module: computerised facial

emotion recognition test

International Affective Picture System

(IAPS) facial emotion recognition test

fMRI evidence of change in neural re-

sponse to emotional stimuli

Bolte 2002

Diagnostic Analysis of Nonverbal Ac-

curacy2 (DANVA2) Faces test: emo-

tion recognition

Solomon 2004

Baghdadli 2013

Reading-the-Mind-in-the-Eyes task:

interpreting mental states from images

of eyes

Fisher 2005

Golan 2006

Cambridge MindReading face-voice

battery: computerised emotion recog-

nition test (close generalisation)

Emotion recognition from novel film

clips (holistic distant generalisation)

Golan 2006

Williams 2012

Matching familiar emotional situa-

tions to familiar facial expressions

(close generalisation)

Matching novel emotional situations

to novel facial expressions but familiar

characters (unfamiliar close generalisa-

tion)

Matching novel emotion situations to

novel facial expressions on novel faces

(distant generalisation)

Golan 2010

Emotion Recognition Test: pho-

tographs of faces

Emotion Vocabulary Comprehension

Test

Ryan 2010

Recognition of emotional expressions

from photographs

Recognition of emotional expressions

from line drawings

Benton Facial Recognition Test (short

form)

Hopkins 2011

NEPSY: Affection Recognition subtest

(recognising emotions from photos of

faces)

Williams 2012

Young 2012




The Faces Task: recognising emotions

from photos of faces

Intervention specific: imitation Motor Imitation Scale: performance

measure of object and gesture imita-

tion

Unstructured Imitation Assessment

Ingersoll 2012

Socially engaged imitation: observed

during examiner/child play session

Landa 2011

Participant behaviour: observation Social Skills Observation: two x 5 min-

utes, during recess or free time in

school

Hopkins 2011

Joint attention and joint engagement

during teacher-child or therapist-child

play

Kaale 2012

Kim 2009

Joint attention and joint engagement

during mother-child play

Kaale 2012

Kasari 2006

Kasari 2010

Schertz 2013

Structured Play Assessment Goods 2013

Wong 2013

Symbolic Play Test Wong 2010

Social skills during classroom observa-

tion

Goods 2013

Wong 2013

Participant behaviour: report Index of Empathy for Children and

Adolescents: self-report

Children’s Social Behaviour Question-

naire (CSBQ): parent report

Begeer 2011

ToM Questionnaire: teacher report Fisher 2005

Social Skills Rating System: parent re-

port

Hopkins 2011

KidScreen, parent-report quality of life

measure

Baghdadli 2013

Problem Behaviour Logs: parent re-

port

Solomon 2004




Ritvo-Freeman Real Life Rating Scale

(RFRLRS): parent report

Wong 2010

PDD-BI social approach, rated by

both parent and teacher

Kim 2009

Vineland Adaptive Behaviour Scales

(VABS)

Schertz 2013

Participant behaviour: direct assess-

ment

Reynell Developmental

Language Scales (post-test; six-month

follow-up, 12-month follow-up)

Kasari 2006

Mullen Scales of Early Learning

(MSEL)

Landa 2011

Schertz 2013

Expressive vocabulary test (five-year

follow-up)

Kasari 2006

Differential Abilities Scale (five-year

follow-up)

Kasari 2006

Test of Problem Solving (executive

function)

Solomon 2004

Acceptibility of Intervention Parent Adherence to Treatment &

Competence: parent report

Caregiver Quality of Involvement

Scale: observational measure during

parent-child play

Kasari 2010

Teacher acceptability of intervention

report

Wong 2013

Children’s Depression Inventory: self-

report

Beck Depression Inventory (BDI) (to

assess parent depression) self-report

Solomon 2004

Parenting Stress Index short form Wong 2010

Economic data None

Unlike the ’Summary of findings’ table and the discussion of intervention effects in the main text, the principal organising element for

this table is the methodology of each outcome measure. This underscores the great difficulty in comparing findings across studies

due to wide variety in assessment scoring systems.



A P P E N D I C E S

Appendix 1. Search strategies

CENTRAL 2013, Issue 7, last searched on 6 August 2013 (35 records)

Previous searches

CENTRAL 2012 (6),10 July 2012 (126 records)

CENTRAL 2010 (3), 2 July 2010 (939 records)

#1MeSH descriptor Child Development Disorders, Pervasive explode all trees

#2autis*

#3asperger*

#4kanner*

#5childhood schizophrenia

#6pervasive developmental disorder*

#7“PDD”

#8language near/3 delay*

#9speech near/3 disorder*

#10(#1 OR #2 OR #3 OR #4 OR #5 OR #6 OR #7 OR #8 OR #9)

Ovid MEDLINE(R), 1946 to July Week 4 2013, last searched on 8 August 2013 (701 records)

Previous searches:

Ovid MEDLINE(R), 1946 to June Week 4 2012, 9 July 2012 (962 records)

Ovid MEDLINE(R) 1950 to July Week 1 2010, 19 July 2010 (4553 records)

1 exp child development disorders, pervasive/

2 autis$.tw.

3 pervasive developmental disorder$.tw.

4 pdd.tw.

5 (language adj3 delay$).tw.

6 (speech adj3 disorder$).tw.

7 childhood schizophrenia.tw.

8 kanner$.tw.

9 asperger$.tw.

10 or/1-9

11 randomized controlled trial.pt.

12 controlled clinical trial.pt.

13 randomi#ed.ab.

14 placebo$.ab.

15 drug therapy.fs.

16 randomly.ab.

17 trial.ab.

18 groups.ab.

19 or/11-18

20 exp animals/ not humans.sh.

21 19 not 20

22 10 and 21

EMBASE (Ovid), 1980 to 2013 Week 31, last searched 6 August 2013 (992 records)

Previous searches:

Embase, 1980 to 2012 Week 27, 9 July 2012 (1979 records)

Embase, 1980 to 2010 Week 28,19 July 2010 (3922 records)

1 exp autism/

2 autis$.tw.

3 asperger$.tw.



4 kanner$.tw.

5 childhood schizophrenia.tw.


7 PDD.tw.

8 language adj3 delay$.tw.

9 speech adj3 disorder$.tw.

10 or/1-9

11 Clinical trial/

12 Randomized controlled trial/

13 Randomization/

14 Single blind procedure/

15 Double blind procedure/

16 Crossover procedure/

17 Placebo/

18 Randomi#ed.tw.

19 RCT.tw.

20 (random$ adj3 (allocat$ or assign$)).tw.

21 randomly.ab.

22 groups.ab.

23 trial.ab.

24 ((singl$ or doubl$ or trebl$ or tripl$) adj3 (blind$ or mask$)).tw.

25 Placebo$.tw.

26 Prospective study/

27 (crossover or cross-over).tw.

28 prospective.tw.

29 or/11-28

30 10 and 29

CINAHLPlus (EBSCOhost) last searched 6 August 2013 (214 records)

Previous searches:

CINAHL 1937 to current, 9 July 2012 (211 records)

CINAHL 1937 to current, 23 July 2010 (847 records)

S26 S15 and 25

S25 S16 or S17 or S18 or S19 or S20 or S21 or S22 or S23 or S24

S24 TI(speech N3 disorder*) OR AB(speech N3 disorder

S23 TI(language N3 delay*) OR AB(language N3 delay*)

S22 TI(childhood schizophrenia*) or AB(childhood schizophrenia)

S21 TI(kanner*) or AB(kanner*)

S20 TI(asperger*) or AB(asperger*)

S19 TI(autis*) or AB(autis

S18 TI(“PDD”) or AB(“PDD”)

S17 TI (pervasive developmental disorder*) or AB (pervasive developmental

disorder*)

S16 (MH “Child Development Disorders, Pervasive+”)

S15 S1 or S2 or S3 or S4 or S5 or S6 or S7 or S8 or S9 or S10 or S11 or

S12 or S13 or S14

S14 TI(effectiv* study or effectiv* research) or AB(effectiv* study or

effectiv* research)

S13 “cross over*”

S12 crossover*

S11 (MH “Crossover Design”)

S10 (tripl* N3 mask*) or (tripl* N3 blind*)

S9 (trebl* N3 mask*) or (trebl* N3 blind*)



S8 (doubl* N3 mask*) or (doubl* N3 blind*)

S7 (singl* N3 mask*) or (singl* N3 blind

S6 (clinic* N3 trial*) or (control* N3 trial*)

S5 (random* N3 allocat* ) or (random* N3 assign*)

S4 randomis* or randomiz*

S3 (MH “Meta Analysis”)

S2 (MH “Clinical Trials+”)

S1 MH random assignment

PsycINFO (Ovid) 1806 to July Week 5 2013 , last searched 6 August 2013 (284 records)

Previous searches:

PsycINFO 1806 to July Week 1 2012, searched 10 July 2012 (1361 records)

PsycINFO searched 2010 via EBSCOhost

1 autism/ or pervasive developmental disorders/ or aspergers syndrome/

2 Autistic Thinking/


4 “pdd”.tw.

5 autis$.tw.

6 asperger$.tw.

7 kanner$.tw.

8 childhood schizophren$.tw.

9 (language adj3 delay$).tw.

10 (speech adj3 disorder$).tw.

11 or/1-10

12 clinical trials/

13 (randomis* or randomiz*).tw.

14 (random$ adj3 (allocat$ or assign$)).tw.

15 ((clinic$ or control$) adj trial$).tw.

16 ((singl$ or doubl$ or trebl$ or tripl$) adj3 (blind$ or mask$)).tw.

17 (crossover$ or “cross over$”).tw.

18 random sampling/

19 Experiment Controls/

20 Placebo/

21 placebo$.tw.

22 exp program evaluation/

23 treatment effectiveness evaluation/

24 ((effectiveness or evaluat$) adj3 (stud$ or research$)).tw.

25 or/12-24

26 11 and 25

PsycINFO (EBSCOhost), searched 23 July 2010 (891 records)

S29 S17 and S28

S28 S18 or S19 or S20 or S21 or S22 or S23 or S24 or S25 or S26 or S27

S27 TI(speech N3 disorder*) OR AB(speech N3 disorder*)

S26 TI(language N3 delay*) OR AB(language N3 delay)

S25 TI(childhood schizophrenia*) or AB(childhood schizophrenia*)

S24 TI(kanner*) or AB(kanner*)

S23 TI(asperger*) or AB(asperger*)

S22 TI(autis*) or AB(autis*)

S21 TI(“PDD”) or AB(“PDD”)

S20 TI (pervasive developmental disorder*) or AB (pervasive developmental

disorder*)



S19 DE “Autistic Thinking

S18 DE ”Pervasive Developmental Disorders“ OR DE ”Aspergers Syndrome“ OR

DE ”Autism“ OR DE ”Rett Syndrome“ Search modes - Boolean/Phrase Interface

S17 S1 or S2 or S3 or S4 or S5 or S6 or S7 or S8 or S9 or S10 or S11 or

S12 or S13 or S14 or S15 or S16

S16 TI (effectiv* research or effectiv* study) or AB (effectiv* research

or effectiv* study)

S15 DE ”Meta Analysis“

S14 TI((clinic* N3 trial*) or (control* N3 trial*)) OR AB ( (clinic* N3

trial*) or (control* N3 trial*))

Database - PsycINFO Display

S12 TI ((tripl* N3 mask*) or (tripl* N3 blind*)) or AB ((tripl* N3 mask*)

or (tripl* N3 blind*))

S11 TI((trebl* N3 mask*) or (trebl* N3 blind*)) or AB((trebl* N3 mask*) or

(trebl* N3 blind*))

S10 TI((doubl* N3 mask*) or (doubl* N3 blind*)) or AB ((doubl* N3 mask*)

or (doubl* N3 blind*))

S9 TI ((singl* N3 mask*) or (singl* N3 blind*)) or AB ((singl* N3 mask*)

or (singl* N3 blind*))

S8 TI((random* N3 allocat* ) or (random* N3 assign*)) or AB((random* N3

allocat* ) or (random* N3 assign*))

S7 TI (randomis* or randomiz*) or AB (randomis* or randomiz*)

S6 DE ”Placebo“

S5 DE ”Mental Health Program Evaluation“

S4 DE ”Psychotherapeutic Outcomes“

S3 DE ”Treatment Outcomes“

S2 DE ”Treatment Effectiveness Evaluation

S1 DE “Random Sampling”

ASSIA (CSA) 1987 to current . Last searched 25 July 2010 (1603 records)

Not available in July 2012 or August 2013

((kw= (randomised or randomized or randomly or random or clinical trial* or control* or placebo* or group* or effectiveness

within 3 research* or evaluat* witin 3 research*)) and((kw=(pervasive developmental disorder* or asperger* or autis* or “pdd”

or kanner* or childhood schizophrenia or language within 3 delay* or speech within 3 disorder*))

Social Services Abstracts(Proquest), 1979 to current, last searched 6 August 2013 (59 records)

Previously searched in 2010 via CSA

(SU.EXACT(“Autism”) OR (“pervasive developmental disorder*” OR asperger* OR autis* OR “pdd” OR kanner* OR “childhood

schizophrenia” OR language NEAR/4 3 delay* OR speech NEAR/3 disorder*)) AND (SU.EXACT(“Random Samples”) OR (ran-

domised OR randomized OR randomly OR random OR clinical trial* OR control* OR placebo* OR group* OR effectiveness NEAR/

3 research* OR evaluat* NEAR/3 research*))

Social Services Abstracts(CSA), 1979 to current, 25 July 2010 (42 records)

((kw= (randomised or randomized or randomly or random or clinical trial* or control* or placebo* or group* or effectiveness

within 3 research* or evaluat* within 3 research*)) and((kw=(pervasive developmental disorder* or asperger* or autis* or “pdd”

or kanner* or childhood schizophrenia or language within 3 delay* or speech within 3 disorder*))

ERIC (Proquest), 1966 to current, last searched 6 August 2013 (284 records)

ERIC 1966 to current, searched 19 July 2012 (461 records)

Previously searched in 2010 via Dialog Datastar

SU.EXACT.EXPLODE(“Asperger Syndrome” OR “Autism” OR “Pervasive Developmental Disorders”) OR “PERVASIVE DEVEL-

OPMENT*

DISORDER*” OR “PDD” OR speech NEAR/3 disorder* OR language NEAR/3 delay* OR autis* OR asperger* OR kanner* OR

“childhood



schizophren*”) AND (SU.EXACT(“Experimental Groups”) OR SU.EXACT(“Control Groups”) OR SU.EXACT(“Followup Studies”)

OR SU.EXACT

(“Longitudinal Studies”) OR SU.EXACT(“Program Effectiveness”) OR ((prospective* OR “follow up” OR evaluat* OR compar* OR

blind*) NEAR/3 stud*) OR (evaluat* NEAR/3 research*) OR ((compar* OR control*) NEAR/3 group*) OR random* OR intervention*

OR

experiment* OR trial*)

ERIC (Dialog Datastar) 1966 to current, searched 21 July 2010 (2982 records)

1.SEARCH:Pervasive-Developmental-Disorders#.DE.

2.SEARCH:pervasive ADJ developmental ADJ disorder$

3.SEARCH:pdd

4.SEARCH:speech NEAR disorder$

5.SEARCH:language NEAR delay$

6.SEARCH:autis$

7.SEARCH:asperger$

8.SEARCH:kanner$

9.SEARCH:childhood ADJ schizophrenia

10.SEARCH:1 OR 2 OR 3 OR 4 OR 5 OR 6 OR 7 OR 8 OR 9

11.SEARCH:Control-Groups.DE.

12.SEARCH:EXPERIMENTAL-GROUPS.DE.

13.SEARCH:LONGITUDINAL-STUDIES.DE.

14.SEARCH:FOLLOW-UP-STUDIES.DE.

15.SEARCH:PROGRAM-EFFECTIVENESS.DE.

16.SEARCH:((prospective$ OR follow ADJ up OR evaluat$ OR compar$ OR blind$) NEAR study).TI,AB.

17.SEARCH:(evaluat$ NEAR research$).TI,AB.

18.SEARCH:((compar$ OR control$) NEAR group$).TI,AB.

19.SEARCH:random$.TI,AB.

20.SEARCH:intervention$.TI,AB.

21.SEARCH:experiment$.TI,AB.

22.SEARCH:trial$.TI,AB.

23.SEARCH:11 OR 12 OR 13 OR 14 OR 15 OR 16 OR 17 OR 18 OR 19 OR 20 OR 21 OR 22

24.SEARCH:10 AND 23

AutismData (http://www.autism.org.uk/autismdata/) last searched 7 August 2013 (59 records)

Searched in 2013 using the phrase “theory of mind”

Previous searches:

10 July 2012 (151 records)


Searched in 2010 and 2012 using the keywords random* or RCT or control* or trial*

ICTRP searched 7 August 2013 (85 records)

Previous searches:


Simple search using terms autis* OR asperg* OR pervasive developmental disorder*

metaRegister of Controlled Trials (mRCT) searched 7 August 2013 (19 records)

Searched in 2013 using the phrase “theory of mind”

Previous searches:


autis* OR asperg* OR pervasive developmental disorder*



C O N T R I B U T I O N S O F A U T H O R S

Draft the protocol S Fletcher-Watson

Develop a search strategy S Fletcher-Watson

Select which trials to include (2 people + 1 arbiter in the event of

dispute)

S Fletcher-Watson, E Manola, F McConnell

Arbiter: H McConachie

Extract data from trials (2 people) S Fletcher-Watson, E Manola, F McConnell

Enter data into RevMan (Cochrane software) S Fletcher-Watson

Carry out the analysis S Fletcher-Watson

Interpret the analysis S Fletcher-Watson, H McConachie

Draft the final review S Fletcher-Watson, H McConachie

Keep the review up to date S Fletcher-Watson

D E C L A R A T I O N S O F I N T E R E S T

A £1000 grant was provided by the charity Research Autism, to pay for some research assistance.

Sue Fletcher-Watson and Helen McConachie - both of these authors have been involved in the development of a therapeutic iPad app

for preschoolers with autism, targeting skills relevant to the current review. An RCT of this app has recently been completed and will

be published in due course. This study may be deemed eligible for inclusion in a future version of this review. The app is now available

on the commercial market and the authors could both receive royalties from sales of the app if these exceed a certain threshold.

Fiona McConnell - none known.

Eirini Manola - none known.

S O U R C E S O F S U P P O R T

Internal sources

• No sources of support supplied



External sources

• Research Autism, UK.

Funding for training and to employ a research associate

D I F F E R E N C E S B E T W E E N P R O T O C O L A N D R E V I E W

Influenced by changes to diagnostic criteria in DSM-5 (APA 2013), and the direction of theory which suggests Theory of Mind (ToM)

primarily underlies social and communication impairments in autism, we removed the domain of FLEXIBILITY AND IMAGINA-

TION from the expected Primary Outcomes.

A further criterion under “Types of Interventions” was implemented to exclude complex, broad-based interventions addressing an array

of social and communication behaviours. While the authors note the efficacy and appropriateness of this approach, these interventions

need to be excluded from our review because they cannot be described to relate directly to the ToM model of autism.

I N D E X T E R M S

Medical Subject Headings (MeSH)

∗Theory of Mind; Child Development Disorders, Pervasive [psychology; ∗therapy]; Emotions; Randomized Controlled Trials as Topic

MeSH check words

Humans



interventions based on the theory of mind cognitive model for …€¦ · t a b l e o f c o n t e n...

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